PLACE IN RETURN BOX to remove this checkout from your record. TO AVOID FINES return on or before date due. DATE DUE DATE DUE DATE DUE 1/88 mm.“ THESIS lo? . I f‘ we THS This is to certify that the thesis entitled The Relationship of Family Functioning to Infant, Parent and Family Environment Outcomes in the First 18 Months Following the Birth of an Infant With Myelodysplasia presented by ' Suzanne Lee Bellinger Feetham. has been accepted towards fulfillment of the requirements for Ph.D. Family Ecology degree in Major professor Date 4/6? (31/30 0-7639 © Copyright by Suzanne Lee Feetham 1 980 THE RELATIONSHIP OF FAMILY FUNCTIONING TO INFANT, PARENT AND FAMILY ENVIRONMENT OUTCOMES IN THE FIRST 18 MONTHS FOLLOWING THE BIRTH OF AN INFANT WITH MYELODYSPLASIA by Suzanne Lee Bellinger Feetham A DISSERTATION Submitted to Michigan State University in partial fulfillment of the requirements for the degree of DOCTOR OF PHILOSOPHY Department of Family Ecology l980 ABSTRACT THE RELATIONSHIP OF FAMILY FUNCTIONING TO INFANT, PARENT AND FAMILY ENVIRONMENT OUTCOMES IN THE FIRST 18 MONTHS FOLLOWING THE BIRTH OF AN INFANT WITH MYELODYSPLASIA By Suzanne Lee Bellinger Feetham The incidence of neural tube defects is l.7 to 3 per l,000 live births in the United States with l30 such births each year in Michigan. Myelodysplasia (also know as myelomeningocele and/or spina bifida) is the most common (60%) of the neural tube defects and is the one defect most frequently associated with survival. Because of improved surgical and medical techniques, increasing numbers of children with this defect are surviving and living with their families. Although myelddysplasia is a serious birth defect, persons with this defect grow to live pro- ductive adult lives. The physical manifestations of myelodysplasia include hydrocephalus in 80% of the children, handicaps of locomotion and continence in 95%, and perceptual and learning disabilities and/or intellect below their unaffected siblings in 25%. The birth of an infant with myelodysplasia affects the family in several ways because the infant requires special care in the home and in both the health care and educational systems. The "cost" of this special care in both energy expended by the family members and in monetary terms is but one effect on the family system. The long term outcome of families raising these children in the home is documented and indicates an increased incidence of divorce, separation, maternal malaise and sib- ling problems over families without children with chronic health prob- lems (Richards & McIntosh, l973; Tew & Laurence, 1973). The conceptual framework selected for this research is the struc- tural—functional approach to family study. Family functioning is Suzanne Lee Bellinger Feetham conceptualized holistically as the activities of everyday life or the way in which the family, as a system, operates across many dimensions. Clinical observations, the review of family theory and the review of re- search related to children with myeldoysplasia and their families suggest there are relationships among infant, parent and family environment variables and family functioning. Therefore, the purpose of this study was to examine the relationships among variables pertaining to the parents, to the infant with myelodysplasia and to the family environment with family functioning at five time intervals in the first l8 months fbllowing the birth of the infant. The dependent variable, family functioning, is a composite score of family functioning across 2l indicators. The independent variables are composite scores of infant, parent and family environment variables as measured by specified items from the Parent Survey (Feetham & Perrin, l977), Profile of Mood States (POMS) (McNair, Lorr & Droppleman, l97l), and infant hospital records and developmental testing using the Early Interven- tion Developmental Profile (Rogers & D'Eugenio, 1977). The subjects for this descriptive longitudinal study were parents and their infants with myelodysplasia admitted to a Myelodysplasia Care Center in a large urban pediatric hospital. The study sample was a total of 38 infants and 66 parents (38 mothers and 28 fathers) who both met the study criteria and agreed to participate. Three hypotheses were tested in this study. Multiple linear regression with stepwise inclusion and listwise deletion was used to test Hypotheses I and II. A simple ANOVA with repeated measures and t tests were used to test Hypothesis III. Suzanne Lee Bellinger Feetham Hypothesis 1, that the infant and parent-family environment pre- dictor variables add significant information to the ability to predict family functioning at 3, 6, l2 and l8 months, was supported by the data. However, Hypothesis II, that the infant composite score variables would account for a greater proportion of variance than the parent variables in predicting family functioning, was not supported. Also, Hypothesis III, that the family functioning discrepant score would be higher at the one year anniversary of the infant's birth than at 3, 6 and l8 months, was not supported by the data. Dedicated to Ann Marie Schraw who, as an infant, introduced me to the world of children with myelodysplasia and their families: for her tenacious commitment for the fullest life and for her family's unrelenting support of her commitment. To her family for sharing their family for my personal and professional growth these last l8 years. Acknowledgments The completion of a dissertation is not done in isolation and with- out collaborative efforts. The original research serving as a basis for this dissertation derived from clinical concerns shared with Jane C. S. Perrin, M. D. As co-principal investigators this Nursing Research Project was supported by the U. S. Public Health Service under grant #NU00632-Ol awarded by the Nursing Research Branch, Division of Nursing, Bureau of Health Manpower, Health Resources Administration, Department of Health and Human Services. The entire research efforts have been supported by research assistants Heather Tweed, R.N., Barbara Jones, R.N., M.S.N., and Sandra Kosik, R.N., M.P.H. The staff of the Center for Health Research, Wayne State Univer- sity, College of Nursing, Betty Walker, Kathy Wiland and Treva McCullough, have been outstanding in proViding consistent, skilled support. Darlene Mood, Ph.D., Sam Agronow, Ph.D., Dave Williams and Gary Moore are recog- nized for their timely and insightful assistance related to the study design and data analysis. In addition, without the openness and willing- ness of the parents to share their time, thoughts and homes, such a study would never be completed. I offer particular recognition to my committee members; Dr. Eileen Earhart, Chairperson, Dr. Linda Nelson, Dr. Beatrice Paolucci, Dr. Hiram Fitzgerald and Dr. Nancy Carlson, who all served as constructive facili- tators throughout my entire doctoral study. To Carolyn Roberts, Rh.D,..R.N., friend and scholar, I thank you for going first, and to my husband, Terry Feetham, I thank you for your waiting, sharing and support. ii TABLE OF CONTENTS LIST OF TABLES LIST OF FIGURES I. INTRODUCTION TO THE STUDY ‘ Statement of the Problem Purpose of the Study Hypotheses Theory Structural functional framework Family functions Assumptions Study Limitations Importance II. LITERATURE REVIEW Independent variables Infant outcomes Etiology of myelodysplasia Clinical manifestations Later outcomes Parent Outcomes Initial response to birth Later outcomes Sibling outcomes Dependent variables Family‘functioning Major areas of family functioning Relationships between the family and broader social units Relationships between the family and subsystems Relationships between the family and the individual Summary 111. METHODOLOGY Design Subjects iii Page vi -l Instruments Infants Early Intervention Development Profile Hospital Records Parents Parent Survey Family Function Survey Profile of Mood States Data Collection Procedures Infants Parents Data Analysis IV. RESULTS OF DATA ANALYSIS Comparison of mothers and fathers responses HypotheSis I Hypothesis II Hypothesis III Summary V. SUMMARY AND CONCLUSIONS Summary Conclu51ons APPENDICES Appendix A Instruments Appendix B Consent Forms Appendix C Table 13 Study Sample BIBLIOGRAPHY iv 89 130 132 133 Table 10 11 12 13 LIST OF TABLES Characteristics of Children with Myelodysplasia Reported in the Literature Compared to Study Sample Study Infant Characteristics Mothers' Characteristics Composite variables Comparison of Mothers' versus Fathers' Responses Profile of Mood States (POMS) Comparison of Mothers' and Fathers' Scores Summary of Stepwise Multiple Regression Analysis to Predict Family Functioning at Time 2 (three months) Summary of Stepwise Multiple Regression Analysis to Predict Family Functioning at Time 3 (six months) Summary of Stepwise Multiple Regression Analysis to Predict Family Functioning at Time 4 (twelve months) Summary of Stepwise Multiple Regression Analysis to Predict Family Functioning at Time 5 (eighteen months) Correlations of Predictor Variables with Family Functioning including the Parent Perinatal Score Change in Mothers' Family Functioning Discrepant Score Between Time Periods Summary of Hypotheses Study Sample Page 41 41 42 66 66 7O 7O 71 71 73 75 76 132 Figure 0501-th LIST OF FIGURES Study Design Independent Variables forming Infant Composite Scores Independent Variables forming Parent Composite Scores Family Functioning Indicators for Dependent Variable Sample Question Format Using Porter Scale Patterns of Responses of Mothers and Fathers on POMS at 3, 6, l2 and I8 monhts vi Page 39 47 49 50 52 67 CHAPTER I INTRODUCTION TO THE STUDY PrOblem The incidence of neural tube defects is 1.7 to 3 per l,000 live births in the United States with l30 such births each year in Michigan. Myelodysplasia (also known as myelomeningocele and/or spina bifida) is the most common (60%) of the neural tube defects and is the one defect most frequently associated with survival. Because of improved surgical and medical techniques, increasing numbers of children with this defect are surviving and living with their families. Although myelodysplasia is a serious birth defect, persons with this defect grow to live productive adult lives. The defect occurs during early embryonic life and by the twenty- eighth day the structural abnormalities are established. Myelodysplasia involves the herniation of the meninges and neural tissue through a spina bifida. A spina bifida, failure of the vertebrae to close, is found in 8% to 20% of the general population and is usually without symp- toms. It is when the meninges and spinal cord also fail to develop that myelodysplasia and its associated problems of hydrocephalus (80%), lower extremity paralysis (95%), and bowel and bladder incontinence (95%), occur. In addition to the physical disabilities of lower extremity paralysis, spinal curvature and skin anesthesia, there is an increased incidence of perceptual and learning disabilities and intellect below their unaffected siblings (25%). The expected life span of these child- ren is full adulthood if they reach adolescence without obesity and major kidney disease (McLaughlin & Shurtleff, l979). The birth of an infant with myelodysplasia affects the family in several ways. An initial and ongoing effect is that several profes- sionals are introduced into the family system, far beyond the usual number introduced with the birth of a normal child. Secondly, the infant requires special care in the home and in both the health care and educational systems. This special care takes more time than the care of a normal infant and often requires the addition of special equipment in the home. The "cost" of this special care in both energy expended by the family members and in monetary terms is a third effect on the family system. The long term outcome of families raising these children in the home is documented and indicates an increased incidence of divorce, separation, maternal malaise and sibling problems over families with- out children with chronic health problems (Richard & McIntosh, 1973; Tew & Laurence, 1973; Walker, Thomas & Russell, 1971). However, the data are usually obtained after the child has been in the home several years and the onset and process for the development of these problems is not documented. In spite of the multiple effects of the birth of an infant with myelodysplasia on the family, the research reports on these families indicate the majority of studies used a univariate linear design and were conducted by members of a single discipline at one time period. The studies have included outcomes focusing primarily on single measures of 1) patterns of parental response to the birth of the infant (Freeston, l97l; Mercer, l974); 2) sibling responses and school behavior (Richard & McIntosh, I973; Walker, Thomas & Russell, l97l); 3) marital integrity (Tew, Laurence, Payne & Rawnsley, I977; Trembath, 1976); 4) family life style changes (Schonell & Watts, I956; Walker, Thomas & Russell, I971); 5) physical and mental health of the mothers (Tew & Laurence, I973; Walker, I970); 6) family functions (Dunlap & HoIIingsworth, I977; Travis, I978); or 7) family planning and sexual relations (Richards & McIntosh, I973; Walker, Thomas, &Russell, l97l). The basis for the traditional linear model in studying families with children with birth defects is Ea; medical model. .”A medical model follows the process of delineation of symptoms, diagnosis and treatment. Etiology has been sought through the study of biology, anatomy and physiology. As researchers in other profeSSions began to study these families they maintained the linear approach of the medical model, but examined a different set of variables resulting in univariate analyses of a very complex multivariate situation. The result of uni- variate research is that pieces of the family, child and/or the handi- cap are examined. In studying the effect of the birth of an infant with myelodysplasia on the family the need is fbr frameworks from which structural-functional characteristics can be examined, while also recognizing the interdependence of the many factors within the family system and between systems interacting with the family. In addition to using univariate designs which do not illustrate interaction among the infant, parent and family environment variables, researchers studying families of children with myelodysplasia have used the mother as the primary data source, have used retrospective data collection and have conducted the studies on non-American popula- tions. When a longitudinal design was used, data collection started 4-5 years following the birth of the child (Tew, Laurence, Payne & Rawnsley,l977). When studied in families with children with myelodysplasia, family functioning is viewed as a variable dependent upon the presence of the child. The interrelationships among parent, infant and family environment variables as they relate to family func- tioning have not been studied. Purpose In recognition of the limitations and results of previous studies of families of children with myelodysplasia, the purpose of this study is to examine the interrelationship among variables pertaining to the parents, to the infant with myelodysplasia and to the family environ- ment as they relate to family functioning at selected time intervals in the first I8 months following the birth of the infant. Knowledge of the patterns of family functioning in the initial period following the birth of the infant and the identification of parent, infant and family environment variables accounting for.the greatest variance in family functioning may be useful to the health practitioner in planning the appropriate times and modes of interventions for these families. This study is part of a larger descriptive study examining both retrospectively and prospectively the outcomes of infants selected for non-surgical treatment of their myelodysplasia versus those infants selected for treatment. The prospective study, begun in I977, includes parent and family environment data in addition to the infant data. This research was supported by the U. S. Public Health Service under grant #NU00632-0I awarded by the Nursing Research Branch, Division of Nursing, Bureau of Health Manpower, Health Resources Administration, Department of Health and Human Services. Hypotheses This research will focus on the effect selected parent, infant and family environment variables have on family functioning in the first 18 months following the birth of an infant with myelodysplasia. Family functioning is conceptualized for the purpose of this study as the parental perception of the degree that family functions are achieved in relation to how much there should be in three areas of relationship: 1) relationships with broader social units, 2) relationships with family subsystems, and 3) relationships with individual members. The difference between what parents perceive there is and should be constitutes a discrepant score, implying parental expectations of family functions are not being met. The research hypotheses are: I) a significant amount of variance in family functioning will be accounted fbr by infant outcome, parent outcome, and family environment variables at each of four time inter- vals (3, 6, 12 and 18 months) following the birth of an infant with myelodysplasia. 2) The composite infant scores including general status, care interventions, and central nervous system status at the neonatal and follow-up period will account fbr greater variance in family function- ing than will parent composite scores of: general status, including marital status, education, health, parent experiences and family en- vironment; perceptions of the perinatal periods; and Profile of Mood State Scores at 3, 6, 12 and 18 months following the birth of an infant with myelodysplasia. 3) Family functioning discrepant scores as measured by parent reports will be greater at the first year anniversary of the birth of the infant with myelodysplasia than at 3, 6 and 18 months following the birth. Theory The conceptual framework selected for this research is the struc- tural-functional approach to family study augmented by concepts from the developmental and family ecological approaches. Also referred to as functional analyses, the structural-functional frameworks for exami- ning family functioning, the dependent variable for this study, are derived from an overall functionalist theory of society, psychology, and social anthropology (Eshelman, 1974). This theoretical origin will be reviewed fbllowed by a discussion of characteristics, limitations and strengths of the structural-functional framework. A brief review of the current status of this framework will be fbllowed by a review of family functions identified by family scholars. The ability to integrate the family structural-functional framework into evolving frameworks such as family development and family ecology supports the relevance of this framework for the study of the effect of the birth of an infant with myelodysplasia on the family in the first 18 months following the birth. The family ecology framework, which recognizes the interdependence of the infant, parent and family environment variables with family functioning, is the basis for the multivariate design of this study (Garbarino, 1977; Morrison, 1974). Eshelman (1974) identifies the structural-functional framework as a dominant theoretical orientation which evolved in the 30's. This framework is commonly used in the study of families because of the overlap of concepts among the structural-functional, developmental and general systems frameworks (Aldous, Osmond & Hicks, 1979; McIntyre, 1966). The Structural—functional framework as used in family research is attributed to several origins. The first is the functionalist branch of psychology, particularly the Gestalt position as it focuses on the relationship between a whole and its parts (Eshelman, 1974). Pitts (1964), however, identifies the origin as the biological sCiences because the use of Aristotelian concepts were insufficient for explaining causality between the parts of living organisms. Two additional possible origins for the structural-functional approach are social anthropology as presented by Malinowski (1939) and Radcliffe-Brown (1935), and social system theorists such as Parsons (1949) and Merton (1968). All of these theorists recognized the interdependence of any aspect of an individual with their setting or larger system. In the application of these theories to the study of families, at times scholars have interpreted the theory out of context which results in a loss of adaptability and relevance of the theory to some research questions (Aldous, Osmond & Hicks, 1979). Structural-Functional Framework Characteristics Several characteristics of the structural-functional framework apply to this research. First, the task of functional analysis is to explain the parts, the relationship between the parts, the relationship between the parts and the whole, and the functions that are performed by, or result from, the relationship formed by the parts (McIntyre, 1966). Review of this relationship between and within systems guides the re- searcher in the identification of variables fbr study. By defining the whole in this study as the.relationship between systems, variables identified for study include family members, the parents, the infant and siblings, and the family environment (e.g., status of the home and community and interaction with the health care system). The structural-functional framework in recognizing system rela- tionships allows for examination of animate (person) variables and inanimate (environmental) variables (Morrison, 1974). This character- istic is important as families with children with myelodysplasia may change their home environment to have adequate space for the special equipment used for the care of the child and/or to live within a con- venient distance to specialized health care and educational facilities (Dunlap & Hollinsworth, 1977). A third characteristic of the structural-functional framework of importance to this study is that the interrelationships and implied interdependence suggest that the groups, systems and behaviors of families and their members are not random and without regularity and predictability (Eshelman, 1974; McIntyre, 1966). Further, Pitts (1964) notes the structural-functional framework should always be capable of showing some reciprocal action. It is therefore anticipated that know- ledge of parent, infant and environmental factors will show a relation- ship with the family's level of functioning. In addition, a fundamental emphasis of this framework is the impor- tance of maintaining the family system. Studies of families of children with myelodysplasia and other conditions resulting in chronic impair- ment indicate these families have a higher incidence of divorce, separa- tion, and maternal malaise (Dorner, I975; Tew & Lawrence, 1973) than families wihtout children with myelodysplasia or other chronic health problems. The system maintenance focus of the structural-functional framework implies that a variety of factors within and outside the family influence the level of functioning of the total unit.' Therefore, in this author's research, variables within and outside the family will be studied. For example, one such measure, leisure activity, is examined as it contributes to the total family system including individual variables of the parents and the infant with myelodysplasia. Family functions The use of family functions is prevalent in research derived from the structural-functional framework. Some researchers define functions of the family by asking the question, "what does it do?", as in actual activities (McIntyre, 1966). Another definition is in terms of the consequences or results of the activities of the family or functional unit. Function is also seen as the contribution an item makes to the maintenance of the whole. Simply stated, the first definition implies the concept of process, the second implies its outcome, and the third implies its content. The primary value of the various definitions of family functions is that they provide general direction to the researcher to consider problems of the relationships between the family system and other societal systems. A limitation of these definitions is that they have limited theoretical weight, therefore leading the researcher into state- ments of generalities and causalities (Pless & Satterwhite, 1973; Smilk- stein, 1978). Family functions as presented by several scholars are presented in Chapter II as a basis for the specific family functions used in this study. 10 Assumptions Several assumptions about the family with an infant with myelo- dysplasia are derived from the structural-functional framework: 1) family functioning is the sum of past experience of family members added to the present combined with future goals and expectations; 2) family functions develop and change over time; 3) family members of all ages act upon and react to their environment; and 4) family functions are influenced by and influence societal expectations, values and norms. Limitations of the Study The principal limitation of this study rests with the sample. It is a convenience sample of 38 families of infants from a total of 46 infants treated in one myelodysplasia care center in a large metropo- litan area in Michigan. The sample is assumed to be representative of the families with infants with myelodysplasia throughout the state, but it may be biased by referral patterns from outstate hospitals into a private, urban hospital. The total sample includes 38 families of which only a limited number supplied the data at each of the time periods. Therefore, the data are analyzed as individual sets at each time period rather than as one set of data with repeated measures over 18 months. The failure to obtain data at each time interval for each family limits the ability to generalize from the data. Incomplete data sets for each family resulted from delayed referral to the myelodysplasia care center, 11 withdrawal from the care center during the 18 month study period and/or inconsistent parental participation in the study. It is not known if the parents volunteering to participate have the same characteristics as those who did not volunteer. A further limitation is having the same informant provide data for both the dependent variable and some independent variables. Also, some data were collected retrospectively, thus subjecting the responses to the effects of cognitive dissonance and poor recall. Importance Of primary importance is that this research derives from a clinical practice question related to variables affecting the family following the birth of an infant with myelodysplasia. The researcher, through clinical experience with these children and their families, can attest to the significance, to clinical practice, of systematic documentation of family functioning fbllowing the birth of an infant with this defect. Another significant feature is that this study provides information not currently available regarding the effect of the birth of an infant with myelodysplasia on its family. Previously researchers have not examined families prOSpectively from the neonatal period and longitudi- nally at several time periods through 18 monhts. In addition, few studies include examination of parents and infants simultaneously. This study includes the examination and discussion of the interrelationships among the infant variables, parent variables and variables in the family environment in relation to their predicting family functioning. A third significant aspect of this study is that data collection includes both 12 the mother and father in contrast to family sociology studies which tend to be comprised only of mothers' perceptions (Safilios-Roths- child, 1970). This is a prospective study from the time of birth of the infant. This is significant because previous studies of these children and their families have been initiated when the child is over five years of age. Although the incidence of divorce, sibling, child care and other family problems are identified in these previous studies, the process leading to these problems can only be hypothesized using retro- spective methodology (Richards & McIntosh, 1973; Tew & Laurence, 1973; Walker, Thomas & Russell, 1971). A review of the pertinent literature related to family functioning, the defect myelodySplasia including the child and outcomes, and the effects on the family is presented in Chapter II. An analysis of fac- tors affecting the response of families to the birth of an infant with myelodysplasia including parent and family environment variables is presented to support the study design and data analysis. CHAPTER II LITERATURE REVIEW Independent Variables Through the review of the literature significant and current con- cepts related to the birth defect myelodysplasia, including the etiology, the effects of this birth defect on the development of the child, and the effects of a child with this birth defect on the family are identi- fied. The concept of family functioning is analyzed to identify those functions pertinent to this research. A model for the study of the interaction of selected parent, infant and family environment variables on family functioning fbllowing the birth of an infant with myelodys- plasia has resulted from this literature review. Infant Outcomes The effects of the birth of a child with myelodysplasia or other chronic diseases on families are frequently reported in the literature. The child with a defect is identified as affecting: employment decisions of parents; living location; vacations and leisure activity; marital relationships; family closeness; siblings, extended family interactions and economic security (Dunlap & Hollinsworth, 1977). In many studies the child with myelodysplasia is identified as the single independent variable determining the dependent variables listed above. However, in addition to specific child variables,'7th‘ere are variables peripheral to the child and variables within the parents and the family environment interacting to influence family functioning (Bronfenbrenner, 1974; Pao- lucci, Hall & Axinn, 1977; Sprey, 1973). 13 14 Variables about the child affecting family functioning include the parent's perception of the neonatal period and the process of being in- formed of the defect, the number and duration of hospitalizations of the infant, and the infant's developmental status (Pricedenham l& Addison, 1978). In this study variables related to the infant are conceptualized as one of the independent variables interrelating with parent and family environmental variables all of which affect family functioning. The infant variables inclusive of the etiology of the defect of myelodys- plasia and its related clinical mainfestations and care are presented first fbllowed by the parent outcomes and family environment variables affecting family functioning. Etiologygof Myelodysplasia The causes of neural tube defects have not been clearly identified. Some investigators support an environmental hypothesis while others support a genetic hypothesis. The environmental etiology is supported by geographic variations and seasonal and annual fluctuations in the frequency of occurrence. Increased risk of occur- rence in siblings after an affected child is born and racial differences support the genetic hypothesis (Carter, 1974; Lorber, 1965). The highest incidence of neural tube defects occurs in the British Isles (Coffee & Jessop, 1957; Record & McKeown, 1949; 1950a; l950b; Smithells, Chinn & Franklin, 1964). The frequency of malfbrmation for this region is estimated to be approximately 3.5 to 10.0 per 1,000 live births (Cziezel & Revesz, I970; Penrose, 1957). In the United States an East-West gradient has been observed with the highest incidence occurring in the New England states and decreasing west of the Rocky Mountains (Alter, 1962; 1963). Descendants of Irish immigrants in Boston have a lower incidence than in Dublin but higher than among 15 non-Irish Bostonians (Naggan & MacMahon, 1967). Over 8,000 infants with neural tube defects are born each year in the United States (Milunsky, 1977). The incidence is higher among the poor (MacMahon, 1970) and among Caucasians. The ratio of girls to boys is about 3:2 (Brockelhurst, 1976). The incidence of the anomaly is usually reported in relation to 1,000 live births which fails to account for a stillbirth rate estimated by Laurence (1969) to be about 25 percent. The overall incidence is computed to be about two per thousand live births. In Michigan, the incidence of neural tube defects is about three per thousand live births (T30 per year), of which 1.7 to 2 (60%) are children with myelodysplasia. The incidence in the British Isles in the last three years has de- clined significantly with the implementation of mass prenatal screening for neural tube defects followed by abortion of the affected fetus (Sunderland & Emery, 1979). The presence of the defect is confirmed by the finding of elevated alpha-feto-proteins in the amniotic fluid and/or maternal serum in the 14th to 17th week of pregnancy (Brockelhurst, 1976). Prenatal diagnosis is available in the United States, but mass screening has only been used for research purposes (Macri, Weiss, Tillitt, Balsam, & Elligers, 1976; Milunsky, 1979). In addition to the regional and ethnic differences in the frequency of occurrence, there seem to be trends of secular and seasonal nature. Gradual decline in the incidence was reported over the period 1940 to 1959 in at least two locations: Scotland and New York. Yet occasional peak years were also observed (Carter, 1965; 1974). Increase in the incidence of births during the winter months, especially in December, as compared to summer, has also been observed. The incidence of 16 myelodysplasia and hydrocephalus relative to maternal age and birth or- der was investigated by Ingalls, Pugh & MacMahon (1954). The greatest incidence fbr Caucasians was observed for birth rank 1, the lowest inci- dence for birth rank 2, and a subsequent gradual increase with increas- ing birth order. The maternal age effect was not detected when birth rank was taken into consideration. The genetic hypothesis is supported by Polman (1950), and Lorber (1965) who suggest a single gene recessive causality. Penrose (1946; 1957) however, suggests that familial incidence is too low to be accounted fbr by a single recessive gene hypothesis and proposes a polygenic hypo- thesis. Unfortunately study of the multiple gene inheritance hypothesis lacks precision and is difficult to substantiate. Preliminary work by Mayeda and Feetham (1974) on eighty-six Michigan families supported a multiple gene hypothesis. Suggested environmental causes for myelodysplasia are agents such as influenza (Sever, 1972); zinc deficiency (Sever & Emanuel, 1972); hormonal pregnancy test (Gal, 1972; Sever, I973); potato blight (Ren- wich, 1973); and softness of local water supply (Fedrick, 1970). Canned meat and tea consumption have also been linked to the occurrence of these defects (Carter, 1974). However, none of these agents have been sufficiently substantiated to be accepted. In the final analysis, the conclusion of genetic predisposition triggered by as yet unknown environ— mental factors, i.e., genetic ang_environmental rather than genetic 9:_ environmental fao’tors,will probably be the answer to causation. The lack of specificity fbr the causation of myelodysplasia is a factor which needs to be taken into account in the study of the effect of an infant with myelodysplasia on the family. Parents identify that 17 their sexual relations are affected by the fear that they could have another child with this defect. In addition families review their pedi- grees for additional evidence of defective genes. Therefore, data on a positive family history for neural tube defects and fetal loss, birth order for the infants with myelodysplasia, and maternal age will be used in constituting the independent variables in this study. Clinical manifestations of myelodysplasia The clinical manifestations of myelodysplasia are evident in several major body systems. These mani- festations will be reviewed from the perspective of their effect both on the child and the family system. Myelodysplasia means failure of nerve development and in this case refers specifically to the spinal cord which is a major structure in the central nervous system. This developmental failure may occur at any point from the cervical (neck) area to the sacral (lower back) area with the most common site being at or below the lumbar region (80%). Since this occurs early in fetal life, further development of the entire central nervous system is altered causing brain and/or brainstem pathology in addition to the spinal cord defect. The level of cord dysplasia and degree of brain malformation determines the degree of paralysis and loss of limb function (95%) and the occurrence of hydro- cephalus in 75-80% (McLaughlin & Shurtleff, 1979). Because the nerves affecting bowel and bladder control originate in the second through fourth sacral segments of the spinal cord, over 95% of the children have bowel and bladder incontinence. Incomplete evacuation evidenced by chronic dribbling of urine and feces is common and increases during periods of crying and activity. Urinary tract complications such as urinary tract infection, ureteral reflux, upper 18 tract dilation and incontinence are also common manifestations (Cass, I976). The outcomes to the family for urological management are the need to express urine manually (crede) from the bladder or to insert a catheter into the bladder to remove the urine; to take the infant for periodic urological examinations; and to monitor the infant for compli- cations. If urinary continence cannot be achieved with catheterization, augmented by medication, or by crede, insertion of an artificial sphinc- ter is now a possibility for a few children. After the age of two the child is placed on a bowel management program to achieve regular timed predictable evacuations. All of these outcomes consume time, energy and increase the costs of raising the infant. In addition, the parents may be distracted from the normal aspects of the infant and focus on the special care. Parents often report that it is hard to find baby- sitters who will perform these care functions in the parents' absence. A primary question from parents of infants with myelodysplasia is the infant's potential fbr ambulation. The most important factors de— termining ambulatory status are level of the lesion, motor power within a given neurosegmental level and the extent and degree of orthopedic defbrmities (DeSouza & Carroll, 1976). Hip dysplasia, deformities of the feet and spinal curvature are the most common orthopedic deformities. In order to achieve ambulation, multiple surgical procedures on the hips and feet, interval casting, splinting and finally bracing are required. In addition, the infant receives physical therapy augmented by parents exercising the infant at home. Prior to braces, special equipment such as standing tables and parapodiums are used. Each aspect of orthopedic intervention requires appointments with health professionals and hospitalization. Concerns which accompany the 19 appointments and hospitalizations are transportation, babysitting for siblings, meals away from home and the loss of work time. As indicated, 85% of these children develop hydrocephalus which is the accumulation of cerebral spinal fluid in the ventricles of the brain. In infants, hydrocephalus is evidenced by the head size increasing faster than normal, enlarged fontanels, developmental delay and cerebral dys- function. The treatment of choice is the surgical insertion of a ventri- cularperitoneal shunt to divert the excess accumulation of cerebral spinal fluid into the abdominal cavity where it is reabsorbed into the body circulatory system (Portnoy & Croissant, 1978; Shurtleff, Foltz & Loeser, 1973). Following insertion of the shunt the children are monitored for the rest of their lives for the clinical evidence of shunt dysfunction. Ideally the original shunt and drainage tubing will function adequately and not become blocked, dislodged and/or infected. The relationship of later cognitive development to the incidence of hydrocephalus is significant, however, reports of the relationship to incidence of shunt dysfunction and the onset of treatment for the dys- function are inconsistent. Tew & Laurence (1974) showed children with hydrocephalus treated by shunts are significantly less intelligent than their normal siblings, whereas children with myelodsyplasia but without hydrocephalus and shunts were the same intelligence as their unaffected siblings. Children with hydrocephalus also scored lower on perceptual- motor functioning than both their siblings and children with myelodys- plasia without hydrocephalus. An inverse relationship existed between level of the lesion and intelligence (Tew & Laurence, 1974). In a study of 173 children,’Soare and Raimondi (1978) reported that 63% with hydrocephalus had 105 over 80 whereas 87% without hydrocephalus had 105 20 over 80. Similar to Tew and Laurence (1974), Soare and Raimondi (1978) found there was no difference between children with myelodysplasia without hydrocephalus and their unaffected siblings. The child's IQ was also related to family income and education. The assumption of intellectual impairment as a consequence of hydrocephalus is probably no longer valid because of the practice of early shunting. Intellectual ability is more likely related to the success of the procedure so that children with repeated episodes of shunt dysfunction and/or infection are at greater risk for intellectual impairment. A deceiving behavior accompanying the hydrocephalus is labeled the "cocktail party syndrome" because of the high level of vacuous verbal activity seen in these children. Unfortunately the high conversational ability of these children masks the inability to conceptualize; hence the behavior is labeled the "cocktail party syndrome." A consequence of this hyperverbal behavior is parental difficulty in accepting a low normal or below normal intelligence score because they mistake verbal ability for IQ. Therefore, due to the known higher incidence of cock- tail syndrome and perceptual problems among children with myelodysplasia, psychometric testing is recommended by age four with comprehensive devel- opmental testing prior to that time (Soare & Raimondi, 1977). However, there is no consistency in the types of tests used to test intelligence and various quotients are used to designate normal vs below normal (Hunt & Holmes, 1975; Lorber & Zachery, 1968). Some authors have used the terms normal and subnormal without reference to intelli- gence quotients. In addition, researchers do not attend to etiological contributions to cognitive development such as brain structure, environ— mental experience and lack of exposure to normal educational settings 21 (Diller, Swinyard & Epstein, 1978). There is general consensus that as the level of the spinal defect ascends above the sacral level, the frequency of hydrocephalus, shunting and mortality increases and cognitive function decreases (Hunt & Holmes, 1975; Lorber & Zachery, 1968; Shurt- leff, Foltz & Loeser, 1973; Soare & Raimondi, 1978, Spain, 1974; Tew, 1977). One secondary manifestation of myelddysplasia is obesity which pro- bably is related to both immobility and overfeeding by solicitous fami- lies. Another secondary manifestation is repeated bone fractures which result from both the immobility and lack of normal nerve innervation and blood supply. Skin breakdown also occurs due to inactivity, poor circu- lation, sensory loss below the level of the lesion, incontinence, casts and braces. Later outcomes In process oriented research it is important to know long term outcomes of these children in order to identify variables for study from infancy. Self care is an important milestone for children with myelddysplasia. However, clinical observations of these children suggest progress toward self care and independence is influenced by several factors. First, logistics and the time required to teach the child to remove and replace braces and then dress over braces and the time for the child actually to perform these tasks result in parents assuming care responsibility beyond the preschool years. Second, often when a child achieves a stage of independence, a hospitalization followed by casting forces the child back to a more dependent role (Steele, 1977). Third, a parent's own need to care for the child may prolong dependence. Mothers are able to identify the child's need for self care but are unable to give this responsibility to the child (Wolfensberger & 22 Menalascine, 1970). Fourth, some children become so obese that they cannot assume self care (Hayes-Allen & Tregg, 1973). In a 1973 study of 1172 families Swinyard, Shakuntala & Nishi- mura (1978), indicated that with extensive therapy and health care, children with myelodysplasia attain independence. Of the 1172 families, whose children had a mean age of 7.8 years, the children had had an average of 2.8 surgeries for shunt revisions, 7.3 kidney x-rays and 1.3 urinary tract infections. As a refléfit of the orthopedic surgeries and physical therapy, 25% ambulated without braces, 43% ambulated with braces, and 31% were mobile in wheelchiars. The longterm outcome of indepen- dence is a serious question in children with myelddysplasia and progress toward self care needs to be assessed from infancy. The outcome for the children born today may be different than for those of the 50's and 60's because of early intervention educational programs, attention to perceptual development from birth and advances in health care (Bates, West & Schmerl, 1977). However, these interven- tions from infancy need to be measured against their effect on the family system as several professionals enter the family system with each inter- vention for the child (Garbarino, 1977; Garbarino & Crouter, 1978). Parent Outcomes The outcomes observed in families of children with myelodysplasia are as important as the specific outcomes of the defect myelddysplasia with regard to the children. In the studies that have been conducted on family response to myelodysplasia, many variables were examined in- cluding immediate effect of the birth on the parents, financial stresses placed on the family, the effect of the handicapped child on siblings, the effect on the marriage and domestic routine, the effect on social 23 life, the physical and mental health of the parents, family planning, and assistance (non-monetary) to the family from relatives and neigh- bors (Hide, Williams & Ellis, 1972). In each instance, the studies were univariate with the single independent variable being existence of a child with myelodysplasia in the home and each of the above factors as theidependent'variable. Initial respgnse to the birth With the birth of any infant the family is altered both structurally and functionally. When a child is born with a defect, providing and securing continued care for the child, the prolonged physical dependence and uncertain future may keep the family in a low state of grief and crisis (Olshansky, 1962). The parents' immediate reactions to the birth of a child with myelodsyplasia are similar to those of other parents of children with defects (D'Arcy, 1968). Most have never heard of the defect and, of those who had heard, few knew of the ways in which the child could be affected. Explanations given by health care personnel were poorly understood by families. Freeston (1971) reports of the 85 parents interviewed, only one quarter of the fathers and mothers felt they under- stood what was explained to them after the birth of the child. Mothers who were separated from their infants by transfers to medical centers felt especially anxious. Fathers reported difficulties immediately after birth in getting back to work and caring for other children while also trying to visit two hospitals and be supportive to their wives (Walker, Thomas & Russell, 1971). The initial period following the birth of a child with a defect is critical in determining the effect of the birth on the family. Families need time to decide what to do after the birth of a child with a defect 24 (Kallop, I973). Apley, Barbour and Westmacott (1967) found that a delay in the treatment of the defect resulted in over-anxiety and disturbance in family patterns in over 50% of the families studied. The information provided during the neonatal period had particular significance on the outcome of the infant. A hopeless prognosis is most frequently associated with the decision not to treat a child with myelodysplasia and the place- ment of the child outside of the home (Hayden, Shurtleff & Broy, 1974; Slobody & Scanlan, 1959; Zachary, 1971). Later Outcomes Contradictory findings have been reported in the literature as to the effect of a child with myelodysplasia on the inte- grity of the marital relationship. Walker, Thomas and Russell reported in 1971 that of the 106 mothers and fathers interviewed, 65% thought their marriage was unchanged by the birth of the myelodysplastic child; 22% felt their marriage had deteriorated. In a study of 278 parents in Michigan, Feetham (1976) reported 23% indicated the child with meylo- dysplasia caused added strain to the marriage, 39% said no difference and 38% indicated they were closer together. Freeston (1971) documents the breakdown of three marriages in the 85 she studied, while Hare and colleagues (1966) noted that, in a great majority of cases, parents said the event had brought them closer together (Hare, Laurence, Payne & Rawnsley, 1966). In contrast to the findings cited above, in a later ten year longitudinal study on the quality of marital relationships, Tew, Payne and Laurence (1974) reported significant deterioration in marital rela- tionships of families of children with major neural tube malformations in England. Fifty-nine families of children with myelodysplasia and 58 25 matched control families were studied. Retrospective perceptions indi- cated that at the time of birth 70% of the index and control families had satisfactory relationships. At the eight to nine year follow-up, only 46% of the index families had satisfactory relationships while 79% of the controls did. This was a statistically significant difference. The divorce rate of the families with affected children was almost two times the national divorce rate, while the controls were below the national average. In 1977, reporting on 56 of the same 59 families, the number of divorces cited was six (10.7%) of which five occurred in the 10 families in which the child was conceived before the marriage. The reported conclusion was that the divorce rate in familiescflichildren with myelodysplasia is "nine times higher than for the local popula- tion" (Tew, Laurence, Payne, & Rawnsley, 1977). These reports are inconsistent and interpretations questionable. One point on which all the researchers agreed was that if a marriage was vulnerable before the birth of the handicapped child, it was more likely to break down than if it were not. MacKeith (1973) notes that the response of parents of children with handicaps is derived from many factors including cultural and social- class attitudes to children in general, but most basically the response is premised on their feelings about having a child with a handicap. Trembath (1976) reports children with myelodysplasia have little adverse effect on family stability if the major factors of medical, social, educational and economic support are present. The effect of a child with myelOdysplasia on family planning and sexual relations has also been studied (Hare, Laurence, Payne & Rawnsley, 1966; Walker, Thomas & Russell, 1971). Freeston (1971) reports that 26 fewer than half of the families she interviewed had sought family plan- ning advice although the majority had decided against having other child- ren. Fifty-nine of the 86 families studied by Richards had had no more children by the time their affected child was two to six years of age. Eleven of these mothers had been sterilized while 32 were employing no active measures to insure against pregnancy. Many of these parents described adverse effects on their sex lives because they did not want "another baby like" their child with myelodysplasia (Richard & McIntosh, I973). The variables of mental and physical health of the parents of children with myelodysplasia have also been studied. Walker, Thomas and Russell (1971) found that onlya few of 106 mothers felt themselves to be “fit and well" while most were "tired, worried and depressed" or substantially worried by their total problem. Half the fathers,ir1com- parison, felt fit and well. In their ten year longitudinal study, Tew and Laurence (1975) measured the mental and physical health of the mothers by using Rutter's Malaise Inventory. The mothers were asked to answer by "yes" or "no" 24 questions referring either to emotional states or to physical states known to have psychological associations. The results indicated that the mothers of children with myelodysplasia had higher stress scores than the mothers of children with psychiatric problems, brain disorder, and physical handicaps whose scores were reported in other studies. Dorner (1975) reported half of the mothers in a sample of 37 families were on medications such as antidepressants. These findings are oon- sistent with Travis' (I976) observation that over time mothers of children with chronic illness become exhausted due to additions in care brought on by the child. 27 The initial responses of parents to the birth of a child with a defect include physical symptoms such as anorexia and fatigue. The parents' sense of emotional well being is labile and based on the state of adjustment to the birth (Wolfensberger & Menalascino, 1970) and the status of the infant (Travis, 1976). Therefore the emotional state of the parent is perceived as a significant variable affecting family functioning. Sibling Outcomes The presence of a handicapped child in a family affects all the members of the family, not only the parents. Several British workers have asked parents to describe the effect the child with myelodysplasia had on other children in the family. Twenty-one percent (17) families interviewed by Richards and McIntosh (1973) felt that the attention they could give the other children was diminished and, there- fore, the myelodysplastic child's presence was detrimental to the sib- lings. Walker, Thomas and Russell (1971) reported a "marked reaction" of a sibling to the myelodysplastic child in 20 of 85 families. In these families the response to the child with the handicap by siblings was marked by excessive negativism or over solicitude. Tew and Laurence (1973), in their longitudinal study of children with myelodysplasia and their families, attempted objective measures of adjustment of siblings. Fifty-nine children with myelodysplasia and their 44 siblings and 59 matched control children and their 63 siblings were studied. Assessing school behavior, the siblings of myelomeningo- cele children showed significantly more maladjustment than the control group. The maladjustment of siblings was greater when the child with myelodysplasia was more mildly handicapped. These findings are supported byMinde, Hackett, Killou and Silver (1972) in their study of siblings. 28 In the Tew and Laurence sample the child with myelodysplasia was treated as the youngest by all family members irrespective of birth order. Family Environment The family environment can be altered following the birth of a child with myelodysplasia. Ways in which family routine is disrupted, causing stressesix>develop, have been reported in the British litera- ture. These were changes in family outings, holidays and domestic rou- tine (Richards & McIntosh, 1973; Schonell & Watts, 1956). Whether parents receive help from friends, neighbors and relatives is often especially important to family functioning in families with children with handicaps. Sixty percent of parents interviewed by Richards and McIntosh (1973) thought that their neighbors and friends had been understanding and helpful after the birth of the affected child. Similarly, those parents studied by Walker, Thomas and Russell (1971) reported receiving help from neighbors in 25% of cases and assis- tance from grandparents in 86% of cases. The type of help sought by mothers include respite from the child to facilitate the mother's mobility for shopping and social activities. The repeated clinical visits and hospitalizations also alter the family environment. Hospitalization of the child was identified as the single most disruptive family event (Freeston, l97l; Tew & Laurence, 1976; Walker, Thomas & Russell, 1971). The combination of special care, clinic visits and hospitalizations affects type of housing, maternal employment, school selection and type of family transportation, while desire for proximity to health care services affects the geographical selection of residence (Dunlap & Hollinsworth, 1977). 29 It is clear from the variables studied and reviewed here that the birth of a child with myelodysplasia affects the outcomes of the children themselves, the parents and the family environment. Some variables may affect the family more than others. Marital harmony, adjustment of siblings, and the physical and mental health of the parents may be crucial outcomes affected. All researchers agree that relation- ships vulnerable to stresses before the birth of a child with myelodys- plasia are in greater peril than those relationships more stable before the birth. DEPENDENT VARIABLE Family Functioning One approach to the study of family functioning is the structural- functional framework which views the family as a social system. Also referred to as functional analysis, the structural-functional framework for examining family functioning is derived from an overall function- alist theory of society, psychology and social anthropology (Eshelman, 1974). An advantage of the structural-functional framework is that it serves to explain the parts, the relationship between the parts, the relationship between the parts and the whole and the functions that are performed by, or result from, the relationship formed by the parts (McIntyre, 1966). A second advantage of the structural-functional framework is that other frameworks, such as family development and family ecology, can be used to augment the framework and expand the parameters of study (Aldous, Osmond, & Hicks, 1979; Bell & Vogel, 1968; McIntyre, 1966). A third advantage is that the structural4FUnctiOna1‘ approach has been effectively applied to the study of the family at several levels from broad macroanalysis to intensive microanalysis. 30 Although the structural-functional framework has only been used in a small proportion of empirical research related to the family, it has had a major effect on family studies. One reason for this major effect is that researchers refer to the importance of family functions even if they do not employ deliberate use of the structural-functional framework (McIntyre, 1966). A second reason is that researchers inte- grate other frameworks into the structural-functional framework (Bell & Vogel, 1968; Levy, 1949). The application of the structural-func- tional framework has served to make family study an integral part of the study of the larger society. Another reason which makes the concepts of the structural-functional framework applicable to a variety of research studies is that there is no single interpretation of family functions. Function may be defined in terms of outcomes, process, and content. Using the concepts of pro- cess, outcome and content, three major areas of functions as relation- ships have been emphasized when applying the structural-functional ap- proach to the study of the family. The areas of family functions when viewed as relationships are: (1) the relationship between the family and broader social units; (2) the relationships between the family and subsystems; and (3) the relationships between the family and the indi- vidual (McIntyre, 1966; Sprey, 1973). Review of scholars' definitions of family functions presents the range of functions from which the specific family functions used in this study were drawn. Functions of the family are seen as patterns of relationships among and between the people and the environment. These functions can be thought of as activities essential to the survival of the family. These functions may be generalized to include tasks such as procreation, 31 socialization, protection, education, and economic concern (Eshelman, 1974). In contrast, Frankena (1970) established a normative set of family functions that are 1) making the lives of each member as good as possible and at the same time, dissuading members from seeking their own good at the expense of others, 2) transmitting patterns of living from one generation to the next generation (education), 3) regulating behavior through discipline based on education not restraint, 4) assisting with achievement of identity for each member, 5) providing a center of leisure for the pursuit of goals, 6) providing a seat of love and emotional gratification, and 7) helping each member build a way of thinking about life, the world, humans, and the universe. Some researchers (Bell and Vogel, 1968) base their descriptions of family functions on the systems model of family exchange using outcome measures. In their model, Bell and Vogel (1968) suggest that the nuclear family trades l) labor for wages, 2) family assets for goods with the economy, 3) loyalty for leadership and compliance, 4) participation for support with the community, 5) adherence for identity with the community, and 6) values and conformity for approval. From this Bell and Vogel (1968) project the following family functions: 1) preparation of goods from the external system for family use, 2) care and maintenance of family possessions, and 3) care of dependent members which includes the socialization process that distributes tasks within the family. On the other hand, Duvall (1971) focuses on the two adult partners and their responsibilities. They are supposed to I) maintain marital ties, 2) maintain effective communication, 3) share in responsibilities, 4) foster the development of the members, 5) provide a safe environment, and 6) protect against the unexpected. 32 Smilkstein (1978), a physician, identifies five family functions from comnon themes in the social science literature. The five functions of adaptation, partnership, growth, affection and resolve were empiri- cally derived to create alfamily function paradigm analogous to the body organ systems. The paradigm acknowledges both a unique component of each function and its interrelationship to the whole. The family functions presented by Gersten (1976) also imply interrelationships between both the individuals and the family and the individuals and the environment. Gersten (1976) identifies that family functions are achieved through a broad range of family behaviors inclusive of: the fulfillment of role functions by family members, the qualities and satisfactions with the marriage, and family communication patterns and processes. Those families which function effectively are seen by Duvall (1971) as successful in narrowing the discrepancies between what might be achieved and what is achieved. The initial discrepancy between the expected birth of a normal child and the birth of a child with a defect precipitates a crisis which disrupts family functioning (Price-Bonham & Addison, 1978; Steele, 1977). When the child continues to be handicapped or chronically ill the family becomes chronically stressed (Martin, H., 1975; Martin, P., 1975; Olshansky, 1962; Waechter, 1977). Family functions become altered in response to this stress in the areas of 1) relationships between the family and broader social units (Tew & Laurence, 1973), 2) the relationships between the family and subsystems (Farber, 1959; Rodgers, 1973; Travis, 1976), and 3) the relationships between the family and the individual (Dorner, 1975; Klein, 1976; Tew, Payne & Laurence, 1974). Family functioning, as the dependent variable for this study, is 33 conceptualized as a composite of the family functions reviewed previously and categorized in the three major areas of relationships. Major Areas of Family Functioning Relationships between the fanfily and broader social units Alterations in the areas of family function of relationships between the family and broader social units seem to emerge from stigma and the limited mobility of the child. Some authors suggest there is a restriction in the range of relationships and activities in families with children with handicaps in response to the stigma of the child, countered by the need to maintain appearances of normalcy to the broader social systems (Travis, 1976; Waechter, 1977). That is, relationships with the extended family, friends and neighbors are altered by the perceived acceptance of the child by the parents (Waechter, 1977). Therefore, social isolation‘ is interpreted by these authors as self-imposed by the family to avoid a perceived stigma. Altered activity patterns are also suggested as a result of the constraints of the handicap (Dorner, l97l; Freeston, 1971; Walker, Thomas & Russell, 1971). In 41 of 94 couples who had not been out socially since the birth of their child, the former group attributed the change in their activities to the extra demands of the care of the child with the handicap (Walker, Thomas & Russell, 1971). The majority of mothers interviewed by Walker and colleagues felt "isolated" as a result of their affected child despite family support (Walker, Thomas & Russell, 1971). In addition, substantial anxiety over leaving the baby with someone else was expressed by two- thirds of the couples. When asked for reasons for joininQEISpina Bifida 34 parent group, 15% of 278 parents cited reduction of isolation as their motivation (Feetham, 1976). In Dorner's (1975) study the parents attri— buted their social isolation to their child's immobility. In a study of 400 rural Alabama families, Dunlap and Hollinsworth (1977) also re- ported a reduction in family activities occurring primarily when the child had a severe physical impairment. Handicapped children without mobility problems were not perceived as changing family relationships. In contrast to the effect of limiting social contacts, the care of a child with myelodysplasia opens the fanfily system to frequent relationships with health professionals. During the neonatal period the family may have contact with over 20 health professionals. Once discharge from the hospital occurs, after the neonatal period, the average number of clinic appointments for myelodysplasia related care is five times during the first year. While parents report satisfaction with the quality of care their child received, they do not find the average health professional supportive (Dorner, 1975; Freeston, l97l; Minde, Hacket, Killou & Silver, 1972). The number of professionals, the unfamiliar terminology and not knowing what is expected, while not feeling comfortable in asking, causes disruption for many parents. In a study by Walker and colleagues (1971), parents reported professionals knew less than they did and therefore tended to be of little help (Walker, Thomas & Russell, 1971). The frequency of hospitalizations also affects the families' rela- tionships to the broader social units. Parents perceive hospitalizations as the single most disruptive event (Dorner, 1975; Freeston, 1971). Walker, Thomas & Russell (1971) reported the mean number of hospitali- zations by age three to be 2.9 in a British sample, In Freeston's (1971) American sample, by age four the mean number of hospital admissions was 6. 35 The presence of a child with a handicap may also affect where the family lives, the type of housing and accessibility to care and educa- tional facilities for the child. All of these factors can alter the family's relationships with the broader society (Dunlap & Hollinsworth, 1977). Relationship between the family and subsystems The second major area of family functioning, i.e., relationships between the family and sub- systems, can also be altered following the birth of a child with myelo- dysplasia. These family functions include the completion of household tasks with or without the help of relatives and the spouse, emotional support from the extended family and overall satisfaction with the marriage. In a study of 107 British families of children with myelodysplasia, 58% of the mothers reported their domestic routine as normal (Walker, Thomas & Russell, 1971). However, 78% of the mothers, who also had normal children, reported the child with myelodysplasia more difficult to raise. In the same study, only 31% of the fathers participated in the ongoing care of the child. However, the families with children with myelodysplasia did receive help and support from the extended family (Klein, 1976; Travis, 1976). Walker, Thomas & Russell (1971) reported the maternal grandmother as most supportive and 26% of the families also received help from the neighbors. In regard to family and child care tasks, Travis (1976) reported the mothers tended to assume the additional responsibilities while the fathers were more apt to abdicate their role. In addition, older siblings either were over solicitous to the child with myelodysplasia or showed excessive negativism (Walker, Thomas & Russell, 1971). 36 The overall quality and satisfaction with the marriage of parents of children with myelodysplasia has been studied through reports of marital breakdown. There are no consistent patterns reported from the studies. In some British studies, the researchers reported the divorce rate to be no higher than the national norm, while one British and two American researchers reported a significant increase in the divorce rate over matched controls (Freeston, l97l; Hare, Laurence, Payne & Rawnsley, 1966; KOIin, Scherzer, New & Garfield, 1971; Richards & McIntosh, 1973). Relationships between the family and the individual The third major area of family functions is relationships between the family and the individual. Specific functions within this area include discussion, disagreements and time with one's spouse, problems and time with child- ren other than the child with myelodysplasia, and satisfaction with sexual relationships. In their study of 400 rural Alabama families, Dunlap & Hollins- worth (1977) reported that 91% of the mothers indicated the handicapped child had no effect on either the marriage or intrafamily relationships. Of the 9% indicating the child had an effect on the marriage and family, 42% reported a positive effect. In addition, 95% of the families re- ported the handicapped child got along well with all immediate and extended family members (Dunlap & Hollinsworth, 1977). The times of the greatest effect of the child with a handicap on the relationships with the family are seen as the initial period fbllow- ing the birth or diagnosis and the first anniversary period (Kallop, 1975; Mercer, 1974; Wolfensberger & Menalascino, 1970). Separation from the infant and the response to the birth both interfere with the 37 normal development of parent-infant relationships. The initial period of multiple decisions and unknown outcomes of the infant add to the altered intrafamily relationships. Clinical observations of these families over time suggest that the parent's perception of the neo- natal period influences their relationships with the infant, other family members and health professionals. Hayden, Shurtleff, and Broy (1974) found placement outside the home to be highly correlated with an initial hopeless prognosis. Summary That some families of children with myelodysplasia and other handicaps can function effectively and adapt to the care of the child while other families experience dysfunction in one or more of the three major areas of family function is evident from the literature. Travis (1976) and Minde (1978) found that the modifications in family function- ing caused by the presence of a child with a handicap are stabilized by the time the child reaches age five. What is important is whether themodifications are positive in relation to expectations of the family functioning. While examples of the effectof the presence of the child with myelodysplasia on specific family functions are reported throughout the literature, data on the interrelationships among parent and infant outcomes and the family environment to the family functioning do not exist. CHAPTER III METHODOLOGY 999.199 The purpose of this descriptive longitudinal study was to examine the interrelationships among infant, parent, and family environment variables to account for the amount of variance in family functioning. The study design is presented in Frigure 1. This study was also explora- tory because questions were raised regarding relationships not studied before or not studied with this population. For example, Garbarino (1977) and Sims, Paolucci & Morris (1972) used designs to study the interrelationships of many variableS'hithe problems of child abuse and malnutrition, but this design is new in the study of children with myelodysplasia and their families. The research hypotheses postulate relationships between 1) infant outcome, 2) parent outcome, and 3) family environment and the discrepancy between expected and actual family functioning through 18 months follow— ing the birth of an infant with myelodysplasia. The dependent variable, family functioning, is a composite score of family functioning across 21 indicators. The independent variables are composite scores of infant outcomes, parent outcomes and the family environment as measured by specified items from the Parent Survey (PS), Profile of Mood States (POMS), and infant hospital records and developmental testing. Subjects The subjects for this descriptive longitudinal study were parents and their infants with myelodsyplasia admitted to a Myelodysplasia Care Center in a large urban pediatric hospital. Forty-six infants with 38 39 TIME PERIOD INFANT MOTHER AND FATHER Neonatal Period Three Months Six Months Twelve Months Eighteen Months Medical History Physical Examination* Echoencephalogram/ ventriculogram Health History and Physical Exam- ination* Early Intervention Developmental Profile (EIDP) Shunt Evaluation** Health History and Physical Exam-' ination EIDP Health History and PhySical Exam- ination EIDP Health History and Physical Exam- ination EIDP Informed Consent Profile of Mood States (POMS) Parent Survey #2 Parent Status Family Environment Family Functioning Prenatal - Form A Parent Status Family Environment Family Functioning Perinatal Perceptions Profile of Mood States (POMS) Parent Survey #3, 4, 5 Parent Status Family Environment Family Functioning Profile of Mood States (POMS) Parent Survey #3, 4, 5 Parent Status Family Environment Family Functioning Profile of Mood States (POMS) Parent Survey #3, 4, 5 Parent Status Family Environment Family Functioning Profile of Mood States (POMS) *Physical examination includes complete neurological examination. .**Radiographic evaluation of shunt function is performed as necessary "at any visit. Figure 1 Study Design 4O myelodysplasia were admitted to the care center during the time of the study from March 1977 to July 1979. These infants represent 40% of the live born infants with myelodysplasia in Michigan in this time period. Criteria for admission of a family to the study were: (1) the infant with myelodySplasia was admitted to the myelodysplasia care center within the first six months following the birth; (2) the infant received medical and/or surgical treatment of the defect; (3) at least one parent was willing to participate in the study; (4) there was continued involvement of the biological parent(s) with the infant; and (5) survival of the infant was anticipated. A total of 38 infants and 66 parents (38 mothers and 28 fathers) comprise the study sample. Eight families were omitted from the study for the following reasons: both of the parents of three infants refused to participate and three parents were not approached on the recommenda- tions of the myelodysplasia center social worker. The reasons given by the social worker for not approaching these three families were because they were non-English speaking; there was severe mental illness in the mother and/or early death of an infant in an already dysfunctional family was anticipated. Two additional families were omitted as the infants were removed from the care of the biological parents. The myelodysplasia care center receives referrals on a statewide basis. The infants and parents in this study came from six counties in southeastern and central Michigan. The comparison of characteristics of the infant sample with those of the population as reported in the literature are shown in Table l. A review of general characteristics is shown in Table 2. 41 Table l Charactistics of Children with Myelodysplasia Reported in the Literature* Compared with Those of the Study Sample (Nt38) Characteristic Previous Reported Study Sample Samples Majority of Conceptions Spring-Summer October-March (75%) Sex ratio: Females:Ma1es 3:2 - 1:1 Lesion at or below L1 80% 91% Presence of hydro- cephalus 75-80% 81% Prepnancy resulting n infant lst lst (23%) *Dekaban, A., 1972; McLaughlin 8 Shurtleff, 1979 Table 2 Study Infant Characteristics (N=38) Characteristic Frequency Percentage Race: Caucasion 28 74% Black 8 21% Hispanic 2 5% First Hospitalization n_ 13 days Birth weight M 3231.706 gms. Head Circumference Percentile* > 90% 15 47% nornel 12 35% < 90% 5 18%+ * 81% were treated for hydrocephalus based on clinical evidence + included preterm and small for date infants 42 All parents (mothers and fathers) of the infants admitted to the myelodysplasia care center were asked to participate in the study unless the center social worker advised against the contact. A total of 66 parents, 38 mothers and 28 fathers, participated in data collection at least once during the time periods. The demographic characteristics of the mothers are listed in Table 3. Marriage was not a criteria for participation; if both parents were available although not married, both the mother and father were asked to participate. Table 3 Mothers' Characteristics (N=38) Characteristic Frequency Percentage Marital Status: Married 29 75 Single 4 13 Single/living with father 3 8 Divorced/Separated 2 4 Education level: < High School 5 13 High School 22 58 1-3 Years Post High School 8 21 Bachelors Degree 3 8 Maternal Age at Birth: < 18 ,years 4 10 19-28 years 23 61 29-34 years 9 24 35 + years 2 5 Pregnancy Planned: Yes 19 50 No 19 50 43 Instruments The infant data are from two primary sources: the hospital records (using chart reviews) and developmental testing, using the Early Intervention Developmental Profile (D'Eugenio & Rogers, 1975). The selection of instruments for infant testing is a significant methodological issue. Medical technology can clarify morphological aspects of brain damage in children with hydrocephalus, but psycho- metric problems persist. The infant is too young for accurate psycho- metric evaluation. The common infant development instruments such as the Gesell or the Bayley Scales of Infant Development are based on sensorimotor development rather than verbal and reasoning skills and are recognized as poor predictors for intelligence (Illingworth, 1971). Tests measuring changes with experience and evolving with age are used reliably only after three years of age. The consequences are that there is a lag between the time of medical assessment of the possible cerebral damage resulting from the myelodysplasia and its associated problems and the ability to assess cognitive function. Also, based on a review of developmental test references, it is clear that instruments that have strong measures of reliability and validity do not exist for assessing children with developmental disabilities. In addition, existing com- prehensive tests of infant development are not designed to provide parents with specific information about their child's development as it relates to the parents' activities with the infant. In light of these considerations, theaEarly Intervention Developmental Profile (EIDP), developed by the Institute for the Study of Mental Retardation and Related Disabilities (ISMRRD) in Ann Arbor, Michigan, was selected for this study. (See Appendix A.1 for instrument.) 44 Early Intervention Developmental Profile The EIDP provides more infant data than do screening tests and does not involve as much time as the Bayley and Gesell instruments, although it comprises many items from these instruments (Honzik, 1976). The advantagesrrfthis instrument are that it is designed for the assessment of infants with developmental disabilities and is combined with a developmental program of interven— tion activities. As a result, following the testing procedure, parents can be given specific activities related to their infant to support their continued development rather than vague ranges of development lacking predictive ability (D'Eugenio & Rogers, 1975). The EIDP is a compilation of major developmental milestones from birth to 36 months of age covering six areas of development. These six areas are perceptual/fine motor, cognition, language, social/emotional, self-care and gross motor. Although testing procedures are included in the EIDP manual, they are not standardized because of the need to modify testing procedures for infants with developmental disabilities. Because of the lack of standardization and age norms, the EIDP is not reconmended for diagnosis but is used for obtaining a developmental age estimate. Professionals from the ISMRDD assessed the concurrent validity of the EIDP by correlating each of the six profile scales with stan- dardized widely used evaluation instruments on 14 children with handi- caps. The correlation coefficients for all scales ranged from a low of .33 between the profile gross motor and Receptive Expressive Emergent Language (REEL) scale and a high of .96 between the profile social scale and cognitive scale and the Bayley Mental Scale. The profile gross motor scale was correlated with both the clinical motor evaluations 45 and the Bayley motor scale with the resulting correlations of .95 and .84 respectively (Rogers & D'Eugenio, 1977). Patnales (1977) compared the EIDP to the Bayley, using a group of normal children, showing a .88 to .98 correlational coefficient between the scales. Interrater reliability was examined by the ISMRRD team by using a tester observer paradigm. The percent of agreement between tester and observers ranged from 80% to 99% with a mean of 89%. Test retest cor- relation coefficients on 15 children at three month intervals ranged from .93 on language at three and six months to .98 on social-emotional measures at three months and six months with all correlations signifi- cant at the p_< .01 levels (Rogers & D'Eugenio, 1977). Hospital Records All other infant data collected from a review of the hOSpital records were determined by a pediatrician and the researcher (a nurse clinical specialist) with a combined experience of over 40 years in the care of children with myelodysplasia. In addition, current research on the short and long term outcomes of these children were re- viewed to assure inclusion of all pertinent data (see Appendix A.2 and A.3 for infant data sheets). Physical examinations, including a complete neurological and developmental assessment, were completed on the infants in the neonatal period, and at 3, 6, 12 and 18 months. Throughout the study infants were not subjected to any procedure and/or examination other than those routinely experienced by all infants treated in the myelodysplasia care center. The neonatal examination delineated the extent of spinal dysra- phism, segmental level of motor and sensory denervation, associated musculoskeletal defects e.g., hip dislocation and club feet, bladder 46 and anal sphincter incompetence and the presence of hydrocephalus (by appearance, head circumference and transillumination). In addition to the physical examination, echoencephalography and/or ventriculography were done in the neonatal period to quantitate ventricular size and brain mantle thickness. A formula published by Shurtleff, Foltz & Loeser (1973) was applied to calculate brain mass in grams which was compared to the brain mass expected for age. Adequate brain mass is a factor in deciding therapy, as Shurtleff's patients with less than 60% normal brain mass preoperatively were all mentally retarded (Shurtleff, Foltz & Loeser, 1973). Follow-up physical examination at 3, 6, 12 and 18 months included assessment of vision and hearing, motor and sensory function and serial echoencephalograms or computerized tomographic (CT) scans as necessary, to determine the degree of hydrocephalus and need for shunt placement or shunt revision. An interval health history also conducted at the follow-up periods included the incidence of acute illnesses and care problems, nutritional history, infant behaviors (such as sleep and socialization), interventions received (such as primary care, immuni- zations and education), the family's response and coping in relation to the infant's birth, and changes in the family environment. All data were recorded on the hospital records by a pediatrician or nurse clinical specialist for later retrieval and analysis. In addition, prenatal, perinatal history and treatment data not obtained in the fellow-up history were also taken from the hospital records. For analysis the individual infant variables were combined into three major composite scores based on the clinical judgment of the inves- tigator and correlates of infant outcomes as identified in the literature 47 (Diller, Swinyard & Epstein, 1978; Laurence, 1976; Lorber, 1971; Richards & McIntosh, 1973; Soare & Raimondi, 1978). The neonatal and infant fbllow-up composite scores included data on general health and developmental status, central nervous system status, and intervention received. The infant fellow-up data were collected at 3, 6, 12 and 18 months. The infant variables used fbr the infant composite scores are presented in Figure 2. Neonatal Infant Score*t ‘ Infant Followup+ General Status Apgar scores care problems associated anomalies total developmental age++ birth weight percentile developmental delay++ gestational age mobility weight percentile Central Nervous System brain mass head circumference percentile head circumference percentile level of motor functioning insertion of ventricular shunt myelodysplasia related problems level of motor functioning shunt status presence of hydrocephalus type of defect Intervention complications acute illness disposition days in hospital length of hospitalization disposition medical treatment emergency room visits number of referrals hospitalizations surgical treatment number of clinic appointments number of surgeries supportive treatment well child care Data source: *Infant neonatal records +Infants hospital records at 3, 6, 12 and 18 months ++EIDP (Early Intervention Developmental Profile) at 3, 6, 12 and 18 months Figure 2 Independent Variables Forming Infant Composite Scores 48 Parents Parent Survey Parent data used for both the independent and dependent variables were obtained from a self-administered questionnaire, the Parent Survey. The Parent Survey (PS), developed by Feetham and Perrin (l977), gathered longitudinal data on a number of facets of parental experiences prior to and following the birth of a child with myelodys- plasia (see Appendix A.4 for Parent Survey). At 3, 6, 12 and 18 months the survey gathered identical data related to (1) general parent status including education, marital status, and perception of physical and mental health, and (2) parent life experiences including the incidence of home relocations and hospitalization, illness and/or the death of friends, relatives, spouse or children. Family environment data collected at these time periods included perception of financial concerns, access to transportation, numbers of persons entering the family system because of the infant with myelodysplasia, a rating of the home environment, and a measure of how and what time was spent with the infant with myelo- dysplasia. The variables comprising the parent and family environment scores are listed in Figure 3. In addition to the Parent Survey, at the 3 month time interval, the parents were asked to complete a questionnaire which included items identicalixithe Parent Survey but were recalled from the time prior to the infant's birth. The parents were also asked their perception of the prenatal and neonatal period. Although the limitations of recall data were recognized, it was not considered appropriate to subject the parents to a lengthy questionnaire during the neonatal period; knowing the confusion and disorientation occurring with the birth of a child with a defect (see Appendix A.5 for Form A - prenatal and neonatal parent data). 49 Parent-Family Environment* educational level marital status perception of mental health perception of physical health Life Experiences death(s) of significant others hospitalization of significant others number of household moves Family Environment Score accessible transportation distance to relatives effect of infant care on finances home environment rating (space, heat, food, safety, clothing) how spend time away from family number of persons living in home number of professionals in home number of professionals seen time spent in activities related to infant Perinatal Family Scoreo family history of neural tube anomalies history of fetal loss maternal age number of pregnancies Prenatal** effect of pregnancy on finances perception of pregnancy prenatal perception of health pregnancy planned Neonatal** distance traveled to see infant expectations of neonatal period expected contact with the infant expected placement of infant information on myelodysplasia information on parent group perception of infant's condition perception of own health in neonatal period Data Source: *Parent Survey at 3, 6, 12, 18 months °Infant neonatal record **Parent Survey Form A at 3 months Figure 3 Independent Variables Forming Parent Composite Scores 50 The Parent Survey was reviewed for face validity by experts in either the care of children with myelodysplasia or family theory. In addition, drafts of the instrument were pretested with several parents of children with myelodysplasia whose children were beyond the age in- cluded in the study. The instruments were-revised following input from all of these sources. Family Function Survey The dependent variable, family functioning, was measured from 21 items included in the Parent Survey. The 21 family function items were developed for this study to measure the three major areas of family functions: (1) the relationship between the family and broader social units, (2) the relationships between the family and sub- systems and (3) the relationships between the family and the individual (McIntyre, 1966). The specific function indicators for family function score are listed in Figure 4. disagreements with spouse talk with friends and relatives discussion of concerns and time in household tasks problems with spouse time in leisure recreational emotional support from friends activities and relatives time miss housework emotional support from spouse time other children miss school help from friends time spent with spouse help from relatives time spouse misses work help from spouse time with children problems with other children time with health professionals satisfaction with marriage time with neighbors satisfaction with sexual relations time you are ill *Data source: Family Functioning Survey Figure 4 Family Functioning Indicators* for Dependent Variable 51 The specific function indicators were derived from review of the family functioning literature (Bell & Vogel, 1968; Duvall, l97l; Eshelman, 1975; Rodgers, 1973) and from the author's 18 years of clinical observa- tions of family functions affected by the birth of an infant with myelodysplasia. The instrument is distinct from other family functioning instruments as it addresses each of the three areas of family functioning and can be used with one or both parents. The instrument includes inter- action with friends, neighbors and relatives as this area of family func- tioning is known to be altered in families with children with handicaps (Tew & Laurence, 1973; 1974; Travis, 1976). Another important aspect of this instrument is that the scoring allows for the computation of a dis- crepant score across all items which is the measure of hypothesis III. Hypothesis III postulates that family functioning discrepant scores as measured by parentsflrrep'Orts 'Iwilllbe greater at the first year anniver- sary of the birth of the infant with myelodysplasia than at 3, 6 and 18 months following the birth. The measures of family function were obtained using the Porter format (Porter, 1962; 1963a; l963b). The Porter format was designed in the early sixties to determine workers' perception of their work situation/environment. The original scale was purported to measure the magnitude, importance, and degrees of need satisfaction of managers in relation to Maslow's (1954) hierarchy of need. The scale was de- signed to measure not only the existing degree of need fulfillment but also the discrepancy between achieved and expected levels in addition to the relative importance of each category (Porter, 1962; l963a; 1963b). 52 While some interpreted the Porter job items as measures of goal attainment (Evans, 1969; Haire, Ghiselli & Porter, 1963), later researchers using the Porter format indicate the stem questions determine the vari- able measured and that the format is adaptable to many conceptual frame- works (Shea, Werley, Rosen & Ager, 1973; Wernimont, Toren & Kappell, 1970). In addition to the management studies, the Porter fbrmat was used in a nationwide survey of faculty and students in health professions in relation to knowledge of family planning (Werley, Ager, Rosen & Shea, 1973), and in a survey of 278 parents regarding expectations of parent groups (Feetham, 1976a; 1976b). The Porter format allows for the computation of a discrepant score for each item indicating perceived expectation for a particular item. Referring to the sample item in Figure 5, the deficiency score represents the difference between ratings on subscales a and b. When the importance score (part c of each item) is used with the discre- pant score both direction and degree of dissatisfaction with the per- ceived existing situation are measured. The importance item (subscale c) can be used as an indication for intervention if the respondant has a high discrepant score (a-b) and high importance score (c). "Amount of time you spend with your other children." a) How much is there now? Little 1 2 3 4 5 6 7 Much b) How much should there be? Little 1 2 3 4 5 6 7 Much c) How important is this to me? Little 1 2 3 4 5 6 7 Much '{discrepant score calculated by(a-b)} Figure 5 Sample Question FOrmat Using Porter Scale 53 The Porter technique is an indirect measure derived from two direct measures by the respondent. According to Porter (1962) this method has two advantages. First, the tendency for a simple response set is reduced as it is more difficult for the respondent to manipu- late the expectation of what is to conform to a sociallygdesirable type of response. This multiple response set also controls for cultural and ethnic diversity as only the discrepant score between the respon- dent's perception of what i§_and should be is used. This is in con- trast to single scale instruments which rely on the researchers' perceived value rating of the "what is" item, i.e., item a (Shaw & Wright, 1967). In addition, this method of measuring expectation or need is considered more conservative than a single question and is a realistic measure in comparing different groups (Porter, 1962). The second ad- vantage is that an indirect measure of satisfaction with the activity inherent in the item is obtained (Porter, 1962). A discrepant score is calculated for each family function item by calculating the difference between the a and b score. Each score is converted to an absolute score as the amount of difference between a and b scores is of significance rather than the direction of the difference. The discrepant score is the amount of agreement between the amount of the activity and the desired amount of the activity. That is, when the amount of the activity is the same as the desired amount, the difference is O. The discrepancy between the amount of reported activity and the amount desired implies the degree of dissatisfaction (Porter, 1962; l963a). Therefbre, the scores closest to 0 indicate the greatest degree of satisfaction with family functioning and those farthest from 0 indicate family functioning is not what it should be as perceived by the 54 respondents. The possible range of total discrepant scores for all the 21 family functioning items is O to 126. Some content validity of the family functioning survey is evi- denced by the significant Pearson correlation coefficients between the Family Functioning Index (FFI) by Pless and Satterwhite (1973) and the Family Functioning Survey used in this study. The Pearson correlations were computed on data from 103 mothers of school children with myelodys- plasia throughout Michigan (r = .54, p < 0.001). The FFI was developed by Pless and colleagues (1972; 1973; 1976) to assess the functioning of families of chronically ill children for the purpose of identifying those children at risk (Pless, Roghmann & Haggerty, 1972; Pless & Satterwhite, 1973; Satterwhite, Zweig, Iker & Pless, 1976). Two types of validation studies, content and construct, have been completed across two samples. In addition, the FFI has evidence of test-retest relia- bility, and a correlation of .72 between the scores of husbands and wives obtained independently (Satterwhite, Zweig, Iker & Pless, 1976). Reliability of the Family Functioning Survey (FFS) was assessed using the alpha reliability coefficient and the coefficient of stability after two weeks using the same sample of 103 mothers in Michigan. The alpha reliability coefficient is 0.72 on 103 subjects and the coefficient of stability after two weeks is 0.85 on 22 subjects. Profile of Mood States The instrument selected to measure the in- dependent variable of emotional response to the birth of the infant with myelodysplasia is the Profile of Mood States (POMS) developed by McNair, Lorr and Droppleman (1971). The POMS is a rapid, economical method of identifying and assessing transient, fluctuating affective states. The POMS, a 65 item factor analytically derived inventory,ii.s a measure 55 of six identifiable mood or affective states: tension-anxiety; depression-rejection; anger-hostility; vigor-activity; fatigue-inertia; confusion-bewilderment. Each of the 65 items is scored on a five point 0-4 scale. (See Appendix A.7 for instrument). The POMS has proved to be a sensitive measure of mood changes over time on psychiatric popula- tions and normal subjects in both experimental and natural situations (Goldberg, 1974; Greenberg, Pillard & Pearlman, 1972; McNair & Lorr, 1964). The same six mood factors are identified, measured reliably and replicated in the research populations whether the rating period is the immediate present or spans a one week period. The POMS was selected for this study as it measures mood states identified by researchers as occurring in parents in response to the birth of a child with a defect. These mood responses include anxiety, depression, anger, fatigue and confusion (Kallop, 1973, Waechter, 1977; Wolfensberger & Menalascino, 1970). According to Mercer (1974) the parents pass through several stages of behavior and mood states in the year following the birth of an infant with a defect, therefore, an instrument measuring fluctuating affective states provides necessary data related to family functioning. The POMS is recommended primarily as a measure of mood states in psychiatric outpatients and as a method for assessing change in such patients. However, it is also recommendeditursimilar purposes on a research basis, for subjects 18 years and older who have had some high school education (McNair, Lorr & Droppleman, 1971). In addition to the six subscores, a total mood disturbance score is obtained by summing the six scores with vigor weighted negatively. The total mood disturbance score is used, as in this study, when a single global estimate of affective state is desired. 56 Use of the POMS showed significant difference in anxiety scores in the same groups over time, when the subjects were exposed to an anxiety provoking experience (Greenberg, Pillard & Pearlman, 1972). There were also significant differences in a small sample of 14 subjects associated with sleep deprivation (Hord, Tracy, Lubin & Johnson, 1975). The POMS was used in several drug related studies on psychiatric patients to test the effects of placebos, nothing and prescribed drugs (Landauer, 1975; Lorr, McNair & Weinsteen, 1964; Raskin & Crook, 1976). The POMS was also used on 36 subjects as a measure of diurinal variations in mood as asserted by self report and verbal content (Taub & Berber, 1974). When a short form of the POMS was used in a study of personality differences between inflammatory bowel disease patients and their healthy siblings, there was no significant difference between the two groups. However, in the same study there was a significant difference between the two groups using a longer test such as the MMPI (McMahon, Schmitt, Patter- son & Rothman, 1973). The POMS has two particular advantages: it is designed to be self-administered to persons with at least a seventh grade education, and it includes a time reference in contrast to many standard personality inventories. By being time referenced the researcher is able to determine whether an enduring personality trait is being measured or the desired mood state (McNair, Lorr & Droppleman, 1971). Extensive reliability and validity testing have been completed on the POMS. For internal consistency the homogeneity of the six replicated POMS factor scores are .87 and above on the 350 male and 650 female psychiatric outpatients. However, the test-retest reliability coeffi- cients are lower (.65 to .70) on 250 psychiatric outpatients. The 57 lower coefficients are considered to be a result of the construct validity of the instrument and the ability to measure mood changes. Factorial validity is evidenced by the six factor analytic repli- cations completed in the development of the POMS as the results were congruent for the different patient and normal samples (McNair, Lorr & Droppleman, 1971). Studies with psychiatric patients receiving therapy versus those not on treatment suggest the scores are not a function of repeated testing but are related to drug and psychiatric treatment (Lorr, McNair & Weinsteen, 1964; McNair, Fisher, Sussman, Droppleman & Kahn, 1970). For concurrent validity the POMS scores were correlated with the Hopkins Symptom Distress Scale (Parloff, Kelman & Frank, 1954), the Taylor Manifest Anxiety Scale (MAS) and the Interpersonal Behavior Inventory (181) using samples of 200 to 1,000 subjects. All correla- tions were significant at the p f .05 or P.: .01 levels. On the symptom distress scales the variance shared by the mood and symptom measures ranged from 5% to 73%. There is a limited effect of demographic data such as sex, education, age, and race on the POMS scores as no more than 5% of the variance is accounted for by these variables (McNair, Lorr & Droppleman, 1971). Also measures of social desirability were found to have low to moderate correlations with the POMS scores of 150 patients. With the exception of the anger subscore (r = .52) the mood scores are independent of role playing and measures of defensiveness or lying (Wiggins, 1964). The parent instruments were organized in the following sequence: demographic, Parent Survey, and at three months, Form A of the Parent Survey (prenatal and neonatal data), and the POMS. The rationale for 58 the sequencing of the instruments was to present material apt to be perceived as most pertinent prior to that probably perceived as least relevant to the respondent. Data Collection Procedures Data were collected from infants and their parents at five time intervals: the neonatal period, 3, 6, 12 and 18 months postpartum. Be- cause of the known perceptual distortions, confusion and shock experi- enced by the parents following the birth of an infant with a defect, only the POMS was administered during the neonatal period (Wolfensberger & Menalascino, 1970). The time intervals for data collection were selected to provide time for family adaptation on the resolution process in response to the birth and for measurable differences in the health and developmental status of the infant to occur. In addition, the first eighteen months is the peak time for the occurrence of morbidity complications and mortality (McLaughlin & Shurtleff, 1979). The data collectors consisted of the principal investigator and one other nurse. Both data collectors were white females between the ages of 32 and 38, with experience in interviewing techniques. In addition, both nurses had experience in working with children with myelodysplasia and their families. The second data collector received an intensive orientation to the data collection protocol and instruments from the principal investigator. Infants The physical examinations in the neonatal period were all completed by a board certified pediatrician specializing in the care of children with neurological problems. The follow up history and physicals (3, 6, 12 and 18 months) were completed by the same pediatrician and/or one of 59 two clinical nurse specialists with master's degrees, one of whom was the researcher. The examinations were conducted during a regularly scheduled appointment at the myelodysplasia care center. During each center visit, the infants were also examined by a urologist, ortho- pedist, neurosurgeon and physical or occupational therapist. Data from each examination were recorded on the medical records and later trans- ferred during chart review to the study infant data sheets for coding and keypunching. Also, data from other clinic visits and hospitalizations were collected during chart reviews. Developmental testing was completed by the two data collectors. The second data collector, who completed the developmental testing on 90% of the subjects, received reliability training on the EIDP by its developers at the Institute for the Study of Mental Retardation and Related Disabilities (ISMRRD) in Ann Arbor, Michigan. Subsequently, interrater reliability between the testers was monitored by using a tester observer paradigm and‘discussions of findings and procedures at periodic intervals. The testing was administered during the regularly scheduled myelodysplasia clinic visit or during home visits. The home visits were conducted if there was insufficient time during clinic, if the infant was not amenable to testing in the clinic, or if the clinic appointment was not kept. All home visits were prearranged at the parent's convenience. Parents Initial Contact Because the research site is a myelodysplasia care center, the practice is for the local hospitals to transfer the infant within hours of birth. As soon as possible after admission of the infant, the parents met with members of the myelOdysplasia care team for 6O discussion of their infant's condition and prognosis. After this dis- cussion, the nurse clinical specialist or the social worker on the myelodysplasia team informed the parents they would be contacted in person by one of the nurse data collectors during a visit to their in- fant. No parent was ever asked to come in just for data collection purposes. The parent(s) were greeted by the data collector, escorted to a waiting room within the patient area and informed of the study (see Appendix B for informed consent). If the parent(s) agreed to partici- pate, written informed consent was obtained and the Profile of Mood States (POMS) administered. If the parent(s) desired, the materials, including the statement explaining the study, were given to the parents with a stamped return envelope. Because of their familiarity with the infant and knowledge of myelodysplasia, the data collectors responded to parent(s) questions about the defect and available resources. Follow-up Contacts The collection of parent data and infant data at 3, 6, 12 and 18 months was completed during a regularly scheduled visit to the myelodysplasia clinic or in the home. With parental agreement, some infants were scheduled one hour before the regularly scheduled afternoon clinic to provide a more relaxed environment for developmental testing and time for parental completion of the questionnaire. If the parents did not complete the questionnaires during the clinic visit and/or both parents were not in clinic, the questionnaires were taken home with a stamped envelope for return. If the clinic appointment was not kept or the developmental testing was not completed during the clinic visit, a home visit was made. In addition, three parents appeared to have difficulty with reading the questionnaires, therefore, home 61 visits were scheduled to permit the data collector to read the ques- tionnaire to the parent. The variety of approaches for data collection were used to limit the amount of missing data and facilitate the parents' participation. An earlier study with parents of children with myelodysplasia in Michigan showed no significant difference between data collected by mail and by home interview (Feetham, 1976). In addition, Bohrnstedt (1967) cites no difference between questionnaire and interview collection of the same data on 200 subjects. Data Analysis All variables from both the infant and parent data were coded such that a low score corresponded to none or a low incidence of a pro- blem or indicated a normal finding and a higher score indicated dys- function. The coding of variables was ordered so that a correlation would be positive if that was expected. Six composite scores were derived from the infant, parent and family environment data for the independent and dependent variables. The process of selection of a variable as part of a composite score was based first on the clinical judgment of the researcher, a nurse clinical specialist, in collaboration with a board certified pediatrician. A second step in developing the composite score was to include variables from research which suggested a correlation between the manifestation of the defect and infant, parent, and/or family environment outcomes. A third step in the process was review of the descriptive statistics for each variable at each time period to assure sufficient data were available for continued analysis. Finally, intercorrelation matrices within each of the six variables were computed. The matrices were 62 examined in order to delete those variables not providing independent information within the composite score for each independent variable. The composite scores used for each independent variable are: (l) the neonatal infant score; (2) the follow-up infant score; (3) the parent and environment score; (4) the parent perinatal score; and (5) the Pro- file of Mood States (POMS) score. In contrast to the composite scores, the POMS score was computed on the 65 items as directed in the test manual. Multiple linear regression was used for hypotheses I and II to determine the relationships between the dependent variable of family functioning and the independent variables of infant, parent and family environment outcomes. Multiple regression is used when analysis of the relationship be- tween a dependent and a set of independent variables is required as in this study design. In addition, the decision to use multiple regression was based on the facts that regression is the procedure of choice when multiple correlations are desired and that this correlation procedure can tolerate violations of parametric assumptions such as a nonrandom sample. Also, the total data sets at each time period, as listed in Appendix C, indicated that a minimum 4:1 or 5:1 subject to variable ratio could be maintained. Because of the complex process in developing the composite scores and the sample size an alpha of p_< .05 was set as the criterion for rejecting the null hypotheses. That a significant amount of variance in family function will be accounted fbr by infant outcome, parent outcome, and family environment variables at each of the four time intervals (3, 6, 12 and 18 months) follow- ing the birth of an infant with myelodysplasia was tested in Hypothesis I. 63 While this hypothesis suggests the importance of the relationships of all the variable scores to family functioning, Hypothesis II postulates the greatest amount of information is provided by the infant scores. One independent variable composite score, the infant neonatal score, remained constant in the regression equation for Hypothesis I at each time. The infant neonatal score was derived from the neonatal period but was retained in the equation to examine data collected at 3, 6, 12 and 18 months because both clinical observation of these families and researchers' reports suggest a carryover effect of the perinatal period on both the infant outcomes and parent outcomes (Farber, 1959; Feetham, 1976; Hayden, Shurtleff & Broy, 1974). Multiple regression analyses on the infant, parent and family environment measures were used to examine the predictive validity of the measures and the amount of variance in family functioning accounted for by each independent variable (composite score). ’rA simple repeated measures ANOVA with control for missing data and t tests were completed to test Hypothesis III: that the family func- tioning score will be greater at the first year anniversary of the birth of the infant than at the other time periods. All of the analyses of the data in this study were computed using programs from the Statistical Package for the Social Sciences (SPSS) and the multiple regression equations were computed using standardized regression coefficients,« ”’/A total of 38 infants and 66 parents comprised the study sample. Of the 66 parents, 38 mothers and 28 fathers participated in data col- lection at least once during the five time periods. Because the sample did not include mother-father pairs for each infant, t tests for indepen- dent samples were computed for both single variables and composite scores 64 to determine if there were significant differences between the responses of the mothers and fathers. CHAPTER IV RESULTS OF DATA ANALYSIS The relationship among the infant, parent and family environment variables with family functioning in families of infants with myelodys- plasia is tested in this research. The dependent variable, family func- tioning, is a composite score of 21 measures of the difference between what the parent perceives a family function should be and what it is. The independent variables of infant, parent and family environment are composite scores derived from review of the infants' hospital records, developmental testing of the infant and the parents' responses on speci- fied items from the Parent Survey. To determine the relationships among the infant, parent and family environment variables and family function- ing, multiple linear regression, t tests, Pearson correlations, and a simple repeated measures ANOVA were used. Comparison of Mothers and Fathers Because the sample did not include mother-father pairs for each infant, t tests for independent samples-were computed for both single wflflufl'- ‘ variables and composite scores to determine if there were significant differences between the responses of the mothers and fathers. There was a total of 66 parents for the 38 infants comprising the study sample. Of these 66 parents, 38 mothers and 28 fathers participated in data collection at least once during the five time periods. There were no significant differences at the .05 level between the mothers' and fathers' responses. The summary of mothers' versus fathers' scores is presented in Table 4 and 5. Because there is no significant difference between the mothers' and fathers' responses, to avoid having two parent scores for some infants, the fathers' data were not used in further analysis. 65 66 Table 4 Composite Variables Comparison of Mothers' versus Fathers' Responses Composite Variable Mothers Fathers Signi- N_ .SQ N_ .SQ t Value ficance Family function _§core , , 3 months 15.550 15.350 11.000 9.426 1.08 NS . 18 months 20.315 21.990 12.000 7.000 1.35 NS Parent-Family Environment 3 months 65.144 4.252 58.053 9.872 0.61 NS 18 months 66.890 9.572 56.240 6.191 2.02 ‘NS POMS 3 months 0.371 0.574 0.339 0.679 0.15 NS 18 months 0.185 0.542 0.264 0.630 —0.30 NS Perinatal Score 28.213 7.908 25.994 7.495 0.83 .fl§ Samgle Size 3 months 18 months* t ers 27 16 Fathers l9 8 *The N at 18 months is affected by the number of infants not yet reaching 18 months of age by the end of the study. Table 5 Profile of Mood States (POMS) Comparison of Mothers' and Father's Scores Time Period Mothers Fathers Combined Mothers vs Signi- N E N Q N Q Fathers fi cance (N) (N) (N) t values 1 (Neonatal) 0.709 01683 0.591 ’0.696 0.660 0.683 0.57 NS (27) (19) (46) 2 (3 months) 0.371 0.574 0.339 0.679 0.345 0.561 0.15 NS (20) (16) (36) 3 (6 months) 0.207 0.609 0.163 0.514 0.193 0.596 0.66 ‘NS (19) ( 9) (28) 4 (12 months) 0.299 0.558 0.055 0.407 0.245 0.516 1.47 NS > (25) (11) (36) 5 (18 months) 0.186 0.542 0.265 0.630 0.212 0.560 -0.30 NS (16) ( 8) (24) 67 Although there is no significant difference between the mothers' and father's scores on both the dependent and independent variables, there are differences in the pattern of the scores. The pattern of responses to the POMS is presented in Figure 6 indicating the fathers' scores are lower than the mothers' until 18 months following the birth when their scores increase over the mothers' scores. In addition, the difference between the parent-family enviornment scores of the mothers and fathers also increased at each time period. The variable POMS 1 I Score .800 . 700 i .600 \ .500 " .\ \ . 400 I \ \\\ .300 “‘ ~ . - .200 \ \ \\ / / \.\\ / \‘\ .100 - \ ‘ 1 l 1 Time 3 6 12 18 Period ‘ Neonatal months months months months Sample Mothers 27) (20) (19) (25) (16) Size Fathers 19) (10) ( 9) (ll) ( 8) ------- Fathers (F) Mothers (M) Figure 6 Patterns of Responses of Mothers and Fathers on Profile of Mood States (POMS) at 3, 6, 12 and 18 months 68 scores contributing to this increasing difference were the infant care score and home environment rating score. The mother's perception of their time in caring for theinfant increased over the fathers' at each time period while their rating of their home environment decreased. A third area of increasing difference in the mothers' and fathers' scores were the family functioning items related to activities and communica- tion with the person assuming the role of spouse with the mothers' discrepant score higher than the fathers'. Between time groups analysis Because the number of mothers completing the Parent Survey over all five time periods was small (N = 4), t tests were computed to determine differences between the groups at each time period. The responses of mothers responding at time two (3 months) but not time three (6 months) were compared; (1) with mothers responding at time f0ur (12 months) and not time two (3 months); as well as (2) compared with mothers responding at both times two (3 months) and four (12 months). The three parent composite scores, parent-family environment, POMS and family function- ing score were used in the t tests. None of the F5 for the t tests were significant at the .05 level. In addition, the scores for the infants of these same sets of mothers were examined. Again, there were no signi- ficant differences between groups on the infant neonatal and fellow-up scores at each time period. The lack of significance on the predictor and criterion variables suggests that subjects with complete data, also subsequently used in the multiple regression, are similar to the subjects not used in the regressions because of incomplete data for all scores. 69 Hypothesis I To test Hypothesis 1, that a significant proportion of variance in family functioning will be accounted for by infant, parent and family environment variables at each of four time intervals (3, 6, 12 and 18 months) following the birth of an infant with myelodysplasia, multiple regression with listwise deletion of variables was used. The stepwise regressions using the four predictor variables at Time 2 (three months) are reported in Table 6. The data fail to show that knowledge of infant, parent and family environment variables adds to the ability to predict family functioning over and above no knowledge. The regression summary for Time 3 (six months) is presented in Table 7. The POMS score was the only variable that contributed significant information to the prediction of family functioning at this time period. The proportion of variance accounted for by the POMS is 60% supporting the research hypothesis. For the regression at Time 4 (twelve months) both infant and parent variables are significant predictors. The variable accounting for the greatest variance (25%) is the neonatal infant vari- able, a score which is unchanged at each time period. The second variable contributing significant infbrmation (accounting f0r 22% of the variance) is again the POMS. The amount of independent variance contributed to the prediction of family functioning by each of these variables was about the same. The analysis summary for Time 4 is presented in Table 8. The POMS variable also is a significant predictor at Time 5 (eigh- teen months) and accounts for 43% of the variance. The neonatal infant score enters the regression equation on the second step but is not sig- nificant. The analysis summary for Time 5 is presented in Table 9. ‘70 mz 8.8.: ¢o_.o mme.c aaa.c acaseoc.>=u sp.e.a-a=oc~a e wm maa.o .Ne.c mmm.c aa~.o Sauces .aoacoaz n ma. .n_.o emo.c mom.o asa.c agate. a::o..oa N mo. 9 on.m_ -~.c ham.c ~a~.° mxoa _ I ll 1 muzoto> oucaa.c.mm.m a a a_ma.m Nu m ccuu.uata anew A__uzv Aegean; x.m. n ms.e on oc.copuucsu x—vsau uu.uosm cu mama—mc< covmmmsomx m—a.up:: umpxaoum we accessm _ a.mo=ooa»: a open» mm sno.o enc.o- oao.c __n.o peace. a::e_.oa 4 mm a~o.e cco.o ~ac.c .om.c mzoa m fl ~85 8.8 896 was 2.3.: .3282 N .mm mem.o ~m_.o- ”mo.o ~m..o u:a:=ac.>=u s..e~a-ucuaaa _ I II I 333...; aa=.a_c.=u.m a L a.ma.m we a Loau.uata aoom Am_.z. Amgucoe omcguv N as.» an mcvcowuucau x—mEeg uu.umca cu mmmxpuc< co.mmacuo¢ o—n.u—:z umwznuum mo auaeeam _ upmaguoa»: m u—auh 71 wm mae.c mmo.c cam.o _a~.o acaecoc_>=u a..s.a-o=otaa . mm m~e._ ..e.o Nem.o mm~.o agate. gaze—Fae n .mm ~mo.. am~.o- amo.o mas.c peace. .aoacaaz N mo. v .a..~ eme.o ewe.o .mo.c mxoa . oucau.cemu.m m. a a_mm_m mm. .m mopaa.ca> Loou.eata aoom Ampuzv .mgaeaz caaogm.u. m each a. a=.=a.ou==a s..saa oo.uaca 3 rubs eovmmfiaoz 253:: omwifim we Dim _ a.m~gooa»: a ape.» mm oo..o Nop.o- mme.o mo~.o acute. aaxa__oa a .wm Nea.c .a..o nae.o ~o~.c acaecagescu a..s.a-u=ucaa n ma. v n.~.m ~v..c ”no.8 .mm.o mzoa N mo. 0 m~o.. ma..c- m.~.c wa..o agate. .aoacooz . aueaa.c.mmam m. c a_mm.m mm. m. mu.a..c.> Loau.uota amen Ac_uzv Amgocoz aspazev e as.» a. ac_=o_oo==a »_.saa ou.uaca 3 mama—22 533.63. 332:: 3:53“... .3 Dim _ m.magooas= m a_aae 72 Although interpretation of the data must be limited because of the sample size available for each regression, it can be noted that the POMS is the major significant predictor of family functioning at three of the four time periods. Although the neonatal infant score is a significant predictor at one time period, neither the parent-family environment score nor the score representing the current status of the infant pro- vide a significant amount of infbrmation at any of the four time periods. The original study design called for inclusion of a fifth predictor variable, the perinatal parent score. However, the number of complete data sets at each time period did not support the inclusion of this fifth variable as the sample having complete data on all variables dropped to nine at six months and four at 18 months. At Time 2 (three months) Pearson correlations of the perinatal variable with the criterion variable, family functioning, suggest that with a larger sample, addition of the parent perinatal variable may add significant information to family functioning. The correlations with the parent-perinatal variable is presented in Table 10. In summary, the data at 6, 12 and 18 months support the research hypothesis that a significant prOportion of variance in family func- tioning is accounted fer by infant, parent and family environment variables. Hypothesis II To test Hypothesis II, thatithe composite infant scores will account for greater variance in family functioning than will the parent-family environment composite score and POMS at 3, 6, 12 and 18 months following the birth of an infant with myelodysplasia, two multiple regressions were computed at each time period. The first multiple regression entered the 73 infant scores into the equation prior to the parent scores. The second mutliple regression reversed the order of entry. Table 10 Correlations of Predictor Variables with Family Functioning Including the Parent Perinatal Score FF2 FF3 FF4 FF5 Sample Size N=l3 N=9 N=ll N= Predictor Variables Parent 0.5499 0.7052 0.4667 0.9956 Perinatal p=0.026* p=0.017* p=0.074 pa0.002** Neonatal 0.1380 0.7189 -0.2857 0.9825 Infant p=0.327 p=0.015* p=0.197 p=0.009** Parent-Family -0.1815 0.1500 0.4047 0.3550 Environ- p=0.276 p=0.350 p=0.108 p=0.322 ment Followup -0.0336 0.0487 -0.2379 0.8516 Infant p=0.457 p=0.451 p=0.24l p=0.074 POMS 0.0071 0.8623 0.3168 0.9845 p=0.49l p=0.001** p=0.17l p=0.008** * p.: .05 ** p 5_.Ol The results of the first regression, infant information entered ahead of parent information, varied by time period. At Time 2 there was no significant change with infant or parent variables. At Time 3 the infant data did not contribute significantly while entry of the first parent variable (POMS) added significant infbrmation. At Time 4 both the infant and parent variables contributed significantly with each accounting fer 25% of the variance. At Time 5 the parent data did not contribute significant information. 74 The results of the second regression, parent information entered ahead of infant information, also varied by time period. Although parent infbrmation was significant at Time 3, Time 4 and Time 5, the infant information did not add significant independent variance at any time. In summary, analysis revealed that the infant variables did not account for significant infbrmation over and above the parent infbrmation or no in- formation in predicting family functioning. The research hypothesis is not supported from these data. Hypothesis III There were only fbur families with complete data at each time period. This negated the use of a repeated measures ANOVA covering all four time periods.:/Therefore, to test Hypothesis III, that family functioning discrepant scores will begreater at the first year anniversary of the birth of the infant with myelodysplasia than at 3, 6, and 18 months fel- lowing the birth, a repeated measures ANOVAMatAB, 6, and 12 months (N=7) and at 6, 12 and 18 months (N=6) was used. The mean scores for these small sample sizes correlated significantly with the means for the total sample. There was no significant difference between the family function- ing scores for each time period fer the mothers with complete data from three to 12 months and mothers with complete data from six to 18 months. In addition, repeated measure t tests were computed between pairs of the time periods on the dependent variable. The dependent variable, family functioning discrepant scores, at 3, 6, and 18 months were each compared with the 12 month family functioning scores. The results of these analyses are presented in Table 11. In summary, the family functioning w-sc:rr1=.15'“ar'e'"not significantly different at the twelve month anniversary period from the 3, 6 and 18 month periods following the birth of the infant. Hypothesis III was not supported by the data. Table 11 75 Hypothesis III Change in Mothers' Family Functioning Discrepant Score Between Time Periods Mothers' Family Functioning Score Sample N_ SQ_ t/F value Signifi- Time . Size cance 2 (three months) 13 18.769 17.801 -0.22 NS_ 4 (twelve months) 19.301 14.688 3 (six months) 11 15.909 11.049 -l.54 .NS 4 (twelve months) 20.818 9.806 4 (twelve months) 12 18.333 133473 -0.48 ‘NS 5 (eighteen months) 20.083 23.083 Mothers at 2 9 14.556 19.749 -0.24 NS_ Mothers at 2 & 4 11 16.364 11.578 Mothers at 4 9 25.778 17.057 1.41 NS_ Mothers at 2 & 4 13 16.846 9.940 2 (three months) 17.714 13.363 3 (six months) 7 14.714 12.244 0.479 NS_ 4 (twelve months) 18.571 .9.360 3 (six months) 15.833 13.512 4 (twelve months) 6 18.500 11.743 0.381 NS 5 (eighteen months) 16.167 12.529 ‘ The family functioning discrepant scores were most stable over time -1' f93_FP9§F mothers with complete data over three or four time periods. Mothers with data far one or two periods had higher family functioning discrepant scores, although there was no significant difference in the -1’ 5. .p". l ‘ . ‘ 02* ~. .2 scores. (When the individual items comprising the family functioning \\ I score are examined the items contributing the greatest increase in the 76 discrepant score are those items related to the relationship with the. person assuming the role of spouse, e.g., emotional support from the f spouse; amount of time with the spouse; and satisfaction with sexual relations. Summary Three hypotheses were tested in this study. The results of the tests of each hypothesis are presented in Table 12. Multiple regression with stepwise inclusion and listwise deletion was used to test Hypotheses I and II. ANOVA with repeated measures and t tests wereused to test Hypothesis III. The incomplete subject data for all four predictor variables and the one criterion variable resulted in a reduction from a potential sample at each time period of 24 to 30 to a sample range of 11 to 16 for the regressions. Table 12 Summary of Hypotheses Hypothesis Research Hypothesis Significance level I Time 2 not supported NS_ Time 3 supported p S .05 Time 4 supported p < .05 Time 5 supported p < .05 11 not supported .NE III. . not supported .NS Hypothesis 1, that parent-family environment and infant data add significant information to the knowledge of family functioning, is sup- ported by the data. However, Hypothesis II, that the infant variables would account fbr a greater proportion of variance than the parent vari- ables in predicting family functioning, is not supported. Also, analysis 77 revealed that Hypothesis III, that the family functioning discrepant score would be higher at the one year anniversary of the infant's birth, is not supported. CHAPTER V SUMMARY AND CONCLUSIONS Summary The incidence of neural tube defects is 1.7 to 3 per 1,000 live births in the United States with 130 such births each year in Michigan. Myelodysplasia (also known as myelomeningocele and/or spina bifida) is the most common (60%) of the neural tube defects and is the one defect most frequently associated with survival. Because of improved surgical and medical techniques, increasing numbers of children with this defect are surviving and living with their families. Although myelodysplasia is a serious birth defect, persons with this defect grow to live pro- ductive adult lives. The physical manifestations of myelodysplasia in- clude hydrocephalus in 80% of the children, handicaps of locomotion and continence in 95%, and perceptual and learning disabilities and/or intellect below their unaffected siblings in 25%. The birth of an infant with myelodysplasia affects the family in several ways. An initial and ongoing effect is that several profes- sionals are introduced into the family system far beyond the usual number introduced with the birth of a normal child. Secondly, the infant requires special care in the home and in both the health care and educational systems. This special care takes more time than the care of a normal infant and often requires the addition of special equipment in the home. The "cost" of this special care in both energy expended by the family members and in monetary terms is a third effect on the family system. The long term outcome of families raising these children in the home is documented and indicates an increased incidence 78 79 of divorce, separation, maternal malaise and sibling problems over families without children with chronic health problems (Richard & McIntosh, 1973; Tew & Laurence, 1973; Walker, Thomas & Russell, 1971). The conceptual framework selected fbr this research is the struc- tural-functional approach to family study (Eshelman, 1974). Family func- tioning is conceptualized holistically as the activities of everyday life or the way in which the family, as a system, operates across many dimensions. Clinical observations, the review of family theory and the review of research related to children with myelodysplasia and their families suggest there are relationships among infant, parent and family environment variables and family functioning (Garbarino, 1977; Travis, 1976). Therefore, the purpose of this study was to examine the rela- tionships among variables pertaining to the parents, to the infant with myelodysplasia and to the family environment with family functioning at selected time intervals in the first 18 months following the birth of the infant. The dependent variable, family functioning, is a composite score of family functioning across 21 indicators. The independent variables are composite scores of infant, parent and family environment variables as measured by specified items from the Parent Survey (Feetham & Perrin, 1977), Profile of Mood States (POMS) (McNair, Lorr & Droppleman, 1971), and infant hospital records and developmental testing using the Early Intervention Developmental Profile (EIDP) (Rogers & D'Eugenio, 1977). Three hypotheses were tested in this study, using multiple linear regression with stepwise inclusion and listwise deletion to test Hypotheses I and II. A simple ANOVA with repeated measures and t tests were used to test Hypothesis III. 80 The subjects for this descriptive longitudinal study were parents and their infants with myelodySplasia admitted to a Myelodysplasia Care Center in a large urban pediatric hospital. Forty-six infants with myelo- dysplasia were admitted to the care center during the time of the study from March 1977 to July 1979. These infants represent 40% of the live 'born infants with myelodysplasia in Michigan in this time period. The study sample was a total of 38 infants and 66 parents (38 mothers and 28 fathers) who both met the study criteria and agreed to participate. (,/There were no significant differences between the mothers' and fathers' ' responses, therefbre, because of unequal N's between mothers and fathers, the fathers' responses were not used in the multiple regression, ANOVA, and t tests to test the three hypotheses. / Hypothesis I, that the infant and parent-family environment predictor variables add significant information to the ability to predict family functioning, was supported by the data. However, Hypothesis II, that the infant composite score variables would account for a greater propor- tion of variance than the parent variables in predicting family func- tioning, was not supported. Also, Hypothesis III, that the family func- tioning discrepant score would be higher at the one year anniversary of the infant's birth than at 3, 6 and 18 months, was not supported by the data. Conclusions Study Design Several considerations for research, clinical practice and teaching derive from this study. The use of a multivariate design to examine family functioning in families with children with myelodysplasia is supported, while raising a challenge to the univariate designs implying single causation between the birth of the child with a defect and the 81 outcomes of the individual or family (Freeston, 1971; Richards & McIn- tosh, 1973; Walker, Thomas & Russell, 1971). The multivariate design is also supported by the work of Garbarino (1977; 1978) and Sims, Paolucci & Morris (1972). If the results of this study can be replicated with other families with children with myelodysplasia or children with other chronic health problems, the applicability of this multivariate design to research, practice, and teaching is strengthened. In addition, as indi- cated previously, this study is unique as it is prospective from the time of birth of the infant and simultaneously examines parent, infant and family environment variables. The value of the prospective design is reinforced by the parent scores which indicate a high level of parental distress in the neonatal period. Structural—functional framework In addition to the use of the multi- variate design being supported, characteristics of the structural-func- tional framework are also supported in this research. The purpose of functional analysis is to explain: 1) the parts, 2) the relationship between the parts and the whole, and 3) the functions that are performed by the parts. The importance of examining the relationships among the parent, infant and family environment variables is reinforced by the fact that different independent variables provided significant information in predicting family functioning at 6, 12 and 18 month time periods. In future studies, other researchers could test other independent variables within the infant, parent and family environment composite scores to determine the amount of information provided by different variables in relation to family functioning. Another characteristic of the structural-functional framework sup- ported by this research is the issue of system maintenance. It is implied 82 in the issue of system maintenance that a variety of factors within and outside the family influence the level of functioning of the total unit. ”The family environment composite score included environmental variables such as availability of transportation, distance to health care services, and the number of persons entering the family system., In future research, a researcher could identify what they perceive to be pertinent infant, parent and family environment variables and test the relationship of these variables to family functioning whether it be with a normal child within a family or a child with a chronic health problem. The ability to iden- tify the significant variables affecting family functioning could provide the basis for the development of clinical assessment tools to be used to provide interventions to prevent family dysfunction. Parent data Other clinical and research implications can be derived from this study. It is important to note that the Profile of Mood States (POMS), a parent measure, entered the regression equation at three of the four time periods at a level providing significant information in predicting family functioning. The POMS scores were highest for the parents at the neonatal period and at the one year anniversary of the birth, supporting clinical observations and researchers' reports of the stress of these periods (Feetham, 1976; Mercer, 1974; Wolfens- berger & Menalascino, 1970). For further interpretation of the POMS data,_studies using larger samples are needed to compare families with normal infants and families with infants with myelodysplasia. If addi- tional studies support the predictive ability of the POMS with family functioning, the POMS could be a useful clinical tool which could aug- ment clinical judgment when planning intervention with families. 83 A, ) Although net__signific1nt, by 18 months following the birth of the infant, the increasing differences between the mothers' and fathers' POMS scores support the need for continued study and suggest the need for preventive intervention. ‘The trend toward differeneesubetween_the mothers and fathers supperts the importance of studying both parentsrf This beginning discrepancy between the mothers and fathers may be the foundation for later family dysfunction. Further data analysis also needs to be completed comparing thePOMS scores of the single mother living with the father, the single mother with no one assuming the spouse role, and the married mother living with her spouse. Neonatal infant data The clinical issuerrfthe extent of the carry- over effect of the neonatal period, following the birth of a child with a defect, to family fUnctioning, is also raised in this study. The independent variable, of the neonatal infant score, presented in the re- gression equation as being most predictive of family functioning at one year following the birth of the infant. By the carryover effect it is implied that the parent's perception of the neonatal period and the actual experiences and outcomes of the infant have an ongoing effect on family functioning. The concept of the carryover effect of the stress of the neonatal period following a birth of a child with a defect is also supported by the clinical observations and research of others (Mercer, 1974; Travis, 1976). Some carryover effect is assumed with the brith of a normal child. However, if the findings of this study were to be replicated with a larger sample of families with infants with myelodysplasia or infants with other defects, yet not replicated with families with normal infants, the extent of carryover of the neonatal period following the birth of an infant with a defect, to later functioning of the family 84 would be supported. If the presence of the carryover effect is supported in future research, the development of instruments to assess parent and infant outcomes that would be predictive of later family functioning should follow the research. Using these data, a clinical approach by the professional, when working with families of children with birth defects, would be to counsel the parents that the one year anniversary of the birth may reactivate thoughts and feelings of the newborn period. Professionals can then provide the family with an opportunity to discuss their feelings and perhaps enable them to progress through another stage in the resolution of the brith of an infant with a defect. Family Environment The family environment items, within the parent- family environment score, were conceptualized as a quantified measure of entry into the family system. Other studies using multivariate models to examine child abuse and nutritional problems in children have also attempted measures of system entry (Garbarino, 1977; Sims, Paolucci, & Morris, 1972). Although the family environment data did not contribute significant infbrmation in the testing of Hypothesis 1, some patterns of the responses are interesting to note and suggest areas for future re- search with both families with normal infants and families with infants with problems. The data are useful from a descriptive standpoint because the extent of system entry in the first 18 months following the birth of an infant with myelodysplasia is documented f0r this sample of 38 families. Indicators of family system entry, in this study, included the num- ber of professionals entering the home system because of the infant with myelodysplasia. This home entry measure included arranging with friends, 85 relatives and/or neighbors for transportation to appointments and documented the number of professionals seen either in the health care system and/or in the home. The home entry score, the parents' perception of their home environment, and time spent in the care of the infant with myelodysplasia were all conceptualized as measures of the family environment. There was no significant difference over time in the home environ- ment score which included an assessment of home: space, heat, food, safety, and clothing. #The family functioning discrepant score increased as themhome score decreased suggesting a relationship between family dysfunction and the perception of the home environment. There were also patterns of reSponse noted in the mothers' perceptions of their care of the infant with myelodysplasia. The patterns in the mothers' data indicated that, as time perceived to be required for special infant care activities increased, the perceived time for worrying also increased, while time for enjoying and playing with the infant decreased. In order to interpret these data fully, data on both the home environment and infant care scores need to be obtained from parents of normal infants and from a larger sample of parents of infants with myelodysplasia. To document entry into the family system, parents reported seeing four to six professionals on each visit to the myelodysplasia care center and averaged 2.4 clinic visits in each time period. In addition, at least one professional entered the home during each time period. These professionals enter the family system because of the infant with myelodysplasia. An area for further study and consideration in planning care intervention is the family's ability to maintain their system boundaries in light of the entry of numerous health professionals (Kantor 86 & Lehr, 1975). An interesting trend in the data was that parents who remained in the myelodysplasia care center from the time of birth of the infant through 18 months or for at least 12 of the 18 months, hid lower ‘family_functioning discrepant scores and lower POMS scores than parents who were in the care center for only three to six months. Further study . is needed to determine if this trend is sustained. The research question' f ' becomes: do families experiencing ongoing comprehensive interdisciplinaryzf/ care function better than families who do not receive this type of care It: or is it that these families who continue with interdisciplinary care have .7’ a higher level of family functioning to begin with and therefore toler-‘r ate multidisciplinary contacts? Future Research Related to Families The state of the art for the development of valid and reliable measures of family functioning as a holistic concept is limited. There are several reasons f0r the limita- tions in this area of research. First, the concept of family functioning ,4. is still vague and poorly defined (Pless & Satterwhite, 1973). As cited in the theoretical discussion, family scholars are inconsistent in both _ , the definitions and measures of family functioning (McIntyre, 1966). if Family functioning may be defined as process, outcome and/or content. The primary value of the various definitions of family functions is that they provide general direction to the researcher to consider problems of the relationships between the family system and other societal systems. AH Onelimfitation of these definitionslis that they have limited theoretical weight and therefore, lead the researcher into statements of generalities and causalities (Smilkstein, 1978). In addition, in reocgnition of the a. complexity of the many conceptual frameworks for family analysis, it would».- be misleading to imply that family functioning can be assessed adequately I," 87 by a simple index. Therefbre, an objective fbr future research related to family functioning should be development of a measure which is of pragmatic value in assisting professionals working with families to identify the potentially dysfunctional family, and also to identify appropriate intervention. A second objective for research related to families is the accurate, systematic documentation of processes used by families to maintain an adequate level of functioning. However, several methodological issues are raised when considering the second objective. The maintenance of both the integrity and boundaries of a family is of primary importance during research. For families responding to unexpected, potentially devastating experiences, such as the subjects of this study, family maintenance must take precedence over data collection. Therefore, the timing and methods of data collection are major issues. Researchers must be sensitive to the context of the family and to evaluate the appro- priateness of extensive interviews, video taping and/or participant observation during the time a family is responding to an unexpected event such as the birth of a child with a defect. Since research related to families with normal children is relatively limited, research on normal families is needed prior to and/or concurrent with research ex- amining family functioning in families responding to unexpected experiences. /”Another research question that needs to be addressed in future studies is focusing on the dyadic interaction of the mother and father, over time, in response to the birth of an infant with a defect.; In addi- tion, outcomes of families receiving care in a multidiscipline compre- hensive care center need to be compared with families receiving, what is perceived by some health professionals as, fragmented care from medical 88 specialists throughout the community. Once the process of response of families to the birth of a child with health problems is documented the ensuing research should test care interventions in a variety of con- texts. In addition to examining the process of response of the family unit to the birth of an infant with health problems, researchers need to study the relationships among and between family members, particularly among siblings. In the preceding conclusions, several implications fer both research and clinical practice derived from this study have been discussed. The need for additional process oriented multivariate research to examine the effect of complex health problems on families is clearly supported. In addition to presenting future research directions in this chapter, the value of this unique study is reinforced as it simultaneously examines parent, infant and family environment variables over the first 18 months following the birth of an infant with myelodysplasia. A difference exists in how families respond to the birth of an infant with a severe defect. This study is a beginning in examining the process of how these families respond. This study provides a model for professionals to understand the process and therefore to assist families to sustain and grow through this experience. APPENDIX A. 89 Appendix A.l Chart review-Neonatal 1 -5 Emily Code Infant Neonatal Assessment Ca rd ‘5 (8) Repair__ Hon-Repair_ (14-17) Oirthdate lo year (28) SGA_W_LGR_ (7) Sex 11 r (ta-22') Birth-eight grams 3 «assert-tarmac: (8 6 9) Race: (23-25) Lengthcm 1 (31-34) at birth 8 (13) Age at Anission (39) Maternal contact visual not incidatad (26-27) Gestational Age _touch __(41-42) Apgar 1 minute none__ _(43-44) 5 minute Level of functioning waeks(35-38)at two weeks (45) Type of defect Heni ngocele Myelomeni ngocele__ RM chesis_ Other—— (46-47) (46-49) Associated anomalies lbtor Sensory ___(67)Hedicalm Treatment —; :2 or G__Topica1 fi 12 53 Rectal prolapse —Systemic 1 5‘ CIUb foot csum 2 55 Hip dysplasia (68) Sirgical Treat- , 56 Cranial/cerebral defects lent ‘ 57 Lacunae Skull None 5 58) GU:uncles. testicle. kid Surgical Back 162 59; :eumgenec bladder _Surgical Head 60 exion contractures ‘ c1713, 61) Orthopedic anmlies 16” 0:11;: SW93? m (62) desnlorphism, FLK. _Intact cord _intact cord microgonathia as no (63 Cardiac E!" ‘5 64 Other neurology (specify): 13 Hydrocephalus yes__ no Conlications: 14 of onset 32 None Rx 20—23) Brain mass ' 35 Post-o Rx 24-25) Ventricular size_ cm (36 CNS (ngn-infection) Rx 26 Ventriculogram/pneunogram yes_ no Fractures Rx % E2§”§§',‘§‘p""°2;“'m __yes___ (38 Heningocele ruptured sac Rx m of fm shu-f—"o (39) Other R! leferalls and consultations (61) Oesposition Fami 1y History 63) 41 Nursing/clinician 52 In Family cm Mother 42 Icamgumry E53; ”gym Foster care_ (64) Father 43 Urology (5.) Other (unchen Wow Nursing home: ' "'— 4; miatrics m—OeEh a '— —- f f ho i 11 - as Social smug. (fllwvfifl A"itvptmt so to m 237 Wye-158601119: at discharge a amlogy _ Primary pEar-e Person Polyhydramu—Psuso yes_ or 49 Genetics (69-68 Maternal 6 A8— 50 Cardiology " History or To tal Io sos— 61 Orthopedics Chart 90 Appendix A.2 review-Infant Follow-Up Family Code Mair: ‘ 5 Repaired Hon-repair Infant Follow-up ___Oalay Repair unths___ days ical s. “'13—— Ione 1. M10 related weigh: mm?"- 2 non. «to «law: Shunt '5 7:; Reason dysfunct t1on No Yes____ repaired Ho_____ ks" Oavelo n: Age Ed” Dela S if areas Cerebral mantle—Iain uss__ ‘m’t o “1;: y Md CIP'CIIFIMCC_______ m1” Specify ONOS asp-ppm s‘fis‘ia Clinic V151??? ‘ u — IW-—— fir scheduled chest —— VCIS__ Mr kept cat ’5‘“ —— Huber disciplines seen/visit— m" —— Specify discipline(s) M” —- Other a oinonents: legs T‘m—ne ventriculogram— lone—'5“ 0"" (39""3'1 Specify service“) PT 106.1 M? __ OT _— flgm Ana- 1 : 09W __ b is': “digit. 1 es :tritionist“ —— ' uros OIS— spgcwy______ Ortho W9 _ mammary Specify Other —- Mculo—skeletal Specify_______ BM Test —- Sensory visual. . — bearing Specify rtive Treatment: Other Specify r y ntervent on__ Analy treated I. [Told related___ Hawington splint: "s__ Ho___ ht nouined___ 2. Hon-nyelo Other (specify) Illnes s: "I. ‘ Mug Care Physician: but noted Type 41:11 .TITTmm-es. etc.) ”"1213! Troa by parents __ 213%), py physician__ 1. MIO'NNW: Otha z, Ion-lyelo Foster—'— care___ m__ related 361119 MC: r c. Ian: 00193633?» ica n 11 mm__ Eurological function level of Type (diaper rash. feeding. lesion “009109.! Lover aim-t? finct no or (urinary tract function (regulation) lane mEd__ Problems (specify) Eer— Inwel OM} wink— pro apse um anilT’m mucus peel?!) 1‘3:"£;muenan_ camp'“N"”'rma'—1'-1TT' o ‘ S italizati s: part Mt "c y 1. 1o ralated____ m3 wmpital , M10 I __ related 91 Appendix A.2 Chart review-Infant Follow-Up Developmental Sumnary m 17. ’1. W? __.. Social tating loilating __ basing _ Cognitive __ Perceptual Fine btor has Ibtor m: 1. nor-l 1. westionable 3. abnorlal hnonal Social __ Fine lbtor We! Gross htor __ 1. 2. 3. I. 5. S. 7. I. 9. non testable 0-2 as. S—S as. H me. 9:41 nos. 12-15 as. “-1! as. til-23 as. 24-27 nos. Dove 27 or other ls there a delay? 1. Yes 2. I: 3. Ihtestable Slant: not applicable Scoring: (1) on age 1m for r is indicated by testing. (2) If child was unable to perfore according to chronological lilestones be was tested at a low age lilestone until le to ss. All eilestone falling beteeen age line and uindica p. were considered failures. (3) lid not test for failure if passed all Illestones an age ea. 92 Appendix A.3 Early Intervention Developmental Profile * [iavelopmantal Programming for Infants and Young Children “ ' “Marlins-mm Early Intervention Developmental Profile by Sally 1. Rogers. Diane B. D'Eugenio. Sara L Brown. Gaul M. Donovan. and Eleanor W. Lynch The University of Michigan Press Amuifimbor REPRINTED WITH PERMISSION 93 Introduction My Intervention Development home is a ompilation of major developmental milestones for use with children whose cognitive. motor. social. self-care. and/or language skills fall within the zero to thiny-six-month developmental range. The profile provides a systematic means of evaluting a child's skills. selecting appropriate objectives for treatment of developmental delays. ad designing an appropriate. individualized curriculum based on a developmental model. The profile evaluates the child's functioning in six areas: perceptual/fine motor. cognition. image. social/emotional. self-care. and gross motor. The profile is designed to be adminstered up to fare times a year for one child by an interdisciplinary team which includes a psychologist or special educator. physical and/or occupational therapist. and a speech therapist. Each section of the gnome assumes a certain degree of disciplinary knowledge and skill on the part of the evaluator. fills which can be taught to other members of the team. Scorim procedures for each item are described in volume I. Assessment and Application. of the threevvolume set Developmental Programming for Infants and Yam Children. Briefly. an item passed by the child is marked with a P if the criteria are met. When there is a question as to whether the child has fully met the criteria for an item. a pass-[ail (PF) should be used to indiate h emergence of the skill measured by that particular item. Clear failures are marked by an F. A final scoring category. 0. is used to signify that an item has been omitted by the evalutor be- one of intervening variables which should be described in the scoring box. The child's performance can be plotted on the profile graph (inside back cover) by marking the ideas: number of consistently passed items in each of the six areas and then connecting the marks. The resulting graph provides a visual representation of the child's relative strengths. weaknesses. and mural developmental level. Objectives for the child in each developmental area can be designed to aid the aquisition of skills in the appropriate developmental level. supporting the strong (highest) tilts as well as facilitating the development of the weak (lowest) area(s). The process of translating ovulation data into individualized programs is fully discussed in volume 1. The “serials necessary to administer the entire profile are listed below: Rattle l2-lnch stick Iell Scull quare box infidel! Twoutsofl-inchardswith four ltd and white pictures of contract: pometric shapes-circle. sci-re. ”on car. com Iottle for doll Penny Doll's chair It; on string Sonar Rains or sugar pellets Inter Sis peas (Jill-inch to lIZ-inch m. use]! (4-inch diameter) diameter) .dlum (loinch diameter) Six-holed pegboard hrge 02-inch diameter) anon Paper-wrapped candy Paper Toy at new: book with cardboard pages Goth or diaper Moises formboard Spoqe lint-end scissors hby bottle M toy Ties identical coffee cups Stake T- l-beh ashes-two and: of sad. m :1 fin. yellow. green. bbck II2~M aibe hhna bum “he” Mi 94 u... Perceptual/Fine Motor Mu”. DEVELOPMENTAL Levers AND ITEMS 3‘” D‘" D‘” 9‘“ 0-2 Monti-s r l I Follows moving object past midline I T I T J 3-5 Honda 2 Integration of grasp reflex 3 Reaches for dangling object 4 Moves head to track moving object 5 Fingers own hands in play at midline 6 Uses ulnar palrnar prehension 7 Looks at hands I Reaches for cube and touches it 9 Uses radial palrmr prehension (uses thumb and two )0 Transfers toy from hand to hand 6-8 Months ll Retains two urbes after third is offered 12 Rakes or scoops up raisin and attains it l3 Has complete thumb opposition on cube M Pulls one peg out of pegboard l5 Uses inferior pincer grasp with raisin 9-ll Months l6 Poker with iaolated index finger I? Drops blocks irnitatively with no pause before release I. Uses neat pincer grasp with raisin l, Attempts to scribble (holds crayon to paper) N Holds crayon adaptively (crayon projects out of radial upoctofthehandmneandupsndoneenddown) mum-mu “y'all!" I“). mrmmnom 95 W“ tun Data Data Data Data DEVELOPMENT "m” “LEVELSANDITDE 1 lz-ls Months Turns of cardboard book Removes cover from small box Places oneortwo in Builds two-cube tower Scribbles (no demonstration) l6-l9 Months 26 Placassixpegsinpegboardwithouthelp Iuilds three-cube tower Places round form in formboard (three forms presented) lmitates myon stroke (myon gripped with butt end thinly in palm) 3:3 20-23 Kendra 30 Haas six pegs in pegboard in 34 seconds 3| flakes vertical and circular scribble after demonstrations 32 Completes threeopiece fcrmbosrd 33 Builds six-cube tower 34 beans to manipulate crayon with fingers 3S Folds paper imitatively 34-27Ionths 36 Drawsveru‘alandhoriacntal sookesimitatively 37 Albutwoormoraeubesforuainmosmokestack im— ZI-Jl Months [nimwwm J J J I 1 32-39 “ooh | 3! Copiesacirclealraadydrawn ] ] L0 Cutswithscisors I “mums-surmoun- Ihl'bt; “ ‘ *, ‘Aep 96 WALLEVEISANDM 0.: Month Uses adaptive movements rather than reflexive reactions T 1 3-5 Hood-s l l Demonstrates vocal contagion Repeats random movements (primary clrcuhr reactions) Watches place where moving obiect disappeand Coordinates two actions in play d-I Months Attains part'nlly hidden object lookstothefloorwhensomethingfalla Uncover: lace Rotatesabottle invertodlosthnn IOO'todrink Acts to have pleasurable interaction repeated 283338 lnInIesaoundsorhandrnovemantsalreadyinhbraper- tone 9-l2 Ionths 52 Pubstrimtoseeureringandsueeeeds $3 lmitates fac'nl movements inesactly gs Attalna completely hidden object (sinus vflbla diplom- ments) 35 t Imperfectly irritates sends and movements nova par- famed before Showsknowlsdgeoftoyhiddenbehindaaaaen l T 37 Routes a bottle inverted l80‘ to drink l3-ll Months Repeatedly finds toy when hidden under one of moral covers (multiple visible displacements) l] J Lss' Ialanoasaight l-lneheubesinacoffeecup J J AA fins—Ion nth-drown“ e - . 1" L "V 97 - lawn Data his Data Dots DEVELOPMENT Nicobar ALLEVELSANDITEMS ‘0 ”has II2-inch cubean l-inchcube cleanly. with pine: gasp lnvertsasmallvialincrdertoretrieveraisin Usaeasticktotrytoattainanobjectoutofrcach 833‘ Uses trial-andoerror approach to precisely irritate new . sounds. words. or movements ”-23 Months Daduceslocationol’obiactfromindlreetviaualcueafui- vbible displacements) Anticipates trajectory by detain-ing around object ea lmitates sounds. words. or body movements immediately and exactly without practicing 67 Indicates knowledge of cause oef feet relationships 24-29 Months latches colored blocks (red. yellow. blue. goon. black) hetendstobeengagedinfarnihraetiviuesMaslaep. uhphmirts) r70 Understands concept of one 7 30-“ Noodle F “It Repeats two digits I 12 latches four shapes (circle. square. star. cross) ldentifiesobiects by their use (as. penny. bottle) mum-armou- hit-0mm” 98 i»... Language "m, DEVELOPMENTAL LEVELSANDITEMS 0'" 0'" De" DI" 0-2 Months E74 I Moves limbs. head. eyes in response to voice. noise 1 J l l J 3.5 Months 75 Vocaliaes when talked to or sung to 16 Turns head in direction of voices and sounds '77 Vocalizcs emotions. intonation patterns 78 Exhibits differentiated crying 64 Months 79 imitates speech sounds '0 Forms bisyllabic repetitions (ma-ma. ba-ba) 9- l 1 Months I Bl Waves or claps when only verbal cue is g'ven I 82 lmitates nonspeech sounds (click. cough) [—83 Inhibits activity in response to no IZ-IS Months .4 Uses appropriate intonation patterns in jargon speech 85 lmitates words inexactly 36 Uses two words meaningfully 87 Uses gestures and other movements to communiate 1649 Months OI Shows body PIN. clothing items. or toys on verbal request 89 Labeb one object 90 Follows two simple directions 9| Uses single words to express wants 92 Points to several body parts (on self or doll) Whlnfimdrmm WW 7&1“. ”Uh lino-Is- 99 lean Numb: DEVELONENTAL LEVELS AND m [73 Names one black and white picture r94 Selects two of three common objects 10-23 Months I 95 I Icg'ns using simple two-word sentences 24-27 Months 96 Uses first name when referring to self 9? Uses three-word sentences 93 Uses negation. no 99 Uses simple pronouns (1. me. you. mine) IN labels at least three common objects or pictures 28-3l Months fiOl Demonstrates an understanding of two prepositions I “)2 Understands body part functions 32-35 Months ")3 Says first and last names 10‘ Demonstrates an understanding of three propositions 10$ Forms questions spontaneously using a verb “)6 Follows two-step commands )0? Forms or uses plurals Whhlsnnurmcnw “twins-”Whom 100 ii... Social/Emotional ”3:” orvzcommnr. trims AND mm D‘“ 0"“ 9‘“ 9'“ 0.2 Months [T08 Quiets when picked up Quiets to face and voice lira Maintains brief periods of eye contact during feeding [iii Smiles or vocalizes to talk and iouch 3-5 Months “2 Reflects adult's smile without verbalizing l l3 Laughs I“ Cries when left alone or put down “5 Shows awareness of strange envsronments ll6 Reaches to familiar people (discriminates strangers) ll? likes physical play I ll Smiles spontaneously ll9 Smiles at image in mirror l2!) Watches adult walk across room 6-9 Months l2l Laughs at pat-a-cake and peek-a-boo games in Withdraws or cries when stranger approaches ”3 Shows dislike when familiar toy is removed 12‘ Pats and touches mirror image 9oll Months l2!) Shows discomfort whenaeparated from motherinstrange environment 126 Participates in pats-cake and peek-s-boo games l2? Repeats voalisations or activity when laughed at l2! Offers toy but does not release Wham-d runawa- W M1100 Wat Fronts 10’) him [Nicobar mummnm l2-l5 Months I129 Offersand releases toy to adult L130 lnitiatesballplayorsocialgamea l6-l9 Month [l3] Usssrnotherassecioebaae.deckingbackwithher Mun!!! 20-23 Months [:32 Oedonally plays near other children [ I33 Oftenclingtoorpushes away adult rl34 Cries when preferred activity is blocked [ l35 Picks up and puts away toys on request 24-21 Months rl36 lodependentlychoosestoyandbegnstcplay [ is? Prefers to play near. but not with. other children I 138 Mimics domestic activities not trains [ I39 IStaresatorpoints to sexual differences 32-35 Month Sepratesfrommothereasilyinstrurgesnviorunsnt | i4i Identifies own sea _L is: Isg’nstonnderstandtakingturns Wyn-hadron.“- Mil-0mm” 102 u... Self-core m" DEVELOPMENTAL LEVELS AND ITEMS 9'" 9'" 9'“ 9'“ Number rm stub 3.5 Months “3 Sucks and swallows pureed foods from spoon I“ Integration of rooting reflex I45 Coordinates sucking. swallowing. and breathing 146 Anticipates feeding with increased activity 147 Gums or mouths pureed food 146 Integration of bite reflex 6-6 Months and swallows textured food lips on spoon to remove from cup with help to up spoon chewing movements motion Holds bottle to drink 9-ll Months Finger feeds small pieces of food Holds cookie Bites cookie cookie Licks food off spoon mashed table drooling l2-lS Months l62 Feeds self with spoon (many spills) I63 Picks up and drinks from cup (some spilling) I64 Chews well l6- l9 Months I65 Drinks from cup without assistance I66 Eats with spoon independently (entire meal) I67 Disaiminates edibles ”-23 Month Ijfl I Unwraps candyzpeels or pits fruit I I I I A “platens-dream mzmmwm 103 sum Item om Due Date Date W Number AL LEVELS AND ITEMS 24-35 Months I69 Begins to use for): I70 Gets drink without help ”I Spoon feeds (no spilling) ‘Tofleflng sum l2- 15 Months I72 Remains dry for l to 2 hour periods I73 Fuses to be changed I6-23 Months I76 Bowel movements are regular I75 Toilet training begins 24-35 Months I76 Uses gestures or words to indicate need to toilet I77 Tofiets independently except for wiping I78 Seldom has bowel accidents Della/Hygiene Skills I2-l5 Months I79 Pulls off bat. socks. or mittens on request I80 Cooperates in diapering and dressing by moving limbs I8l Attempts to brush hair I649 Months I82 Imitates simple grooming actions with various objects. i.e., toothbrush. comb. washcloth. with little attempt to acorn , 217-23 Months I I83 Undresaes completely except for fastenings [ I84 Attempts to put shoes on [ I85 Unzips and zips large zipper 34.3] Months [m Puts on simple clothes without assistance (hat. pants. shoes, etc.) L m IWashesanddrieshandswithaasistance 7 32-35 Months [ I88 Dries hands independently l I89 Puts on cost. dress. T‘shirt except for buttoning L I90 Undoea large buttons. snaps. shoelaces deliberately luau-1mm memuimm Whit 104 a... Gross Motor ”M". DEVELOPMENTAL LEVEisANDri-EMS 9‘" D‘" 9‘“ D'“ 0-2 Months . turns head to either side Neck r'uhting Upright: head bobs but stays erect Upright‘ negative support reaction (integration of step ping reflex) . kicks feet alternately 3-5 Months I99 Inteyation of Moro reflex zoo Prone:headandchestareraised to90'with forearm ”PM 201 Upright: bears small fraction of weight on feet ”2 Prone: props with extended arms 203 Pulled to sitting with no head lag 20‘ Pulls self to sitting 20$ Prone: rolls to supine 206 hone; integration of TLR 207 Supine: intep-ation of T”! 208 Mi integration of STNR 209 Supine: intention of ATNR 6-8 Months 2I0 Sitting; trunk erect in chair 2| l Upright: extends legs and takes large fraction of weight 12— Sits alone for at least 5 seconds 2I3 Supine: lifts head spontaneously 2M Inteuation of neck righting Miriam“ You-M rfiuzmmwhp 105'- I.“ M DEVELOPMENTAL LEVELS mo mans 1"" 9‘" 9‘" Number 2IS Iody on body righting begins 2I6 Supine: labyrinthine righting 217 Supine: optial righting 2I8 hone: Landau reflex 2I9 Sitting: protective extension to the front 220 Parachute reaction 22I Prone and supine: equilibrium reactions 222 Sitting: labyrinthine righting when tipped to sides 223 Sitting: optical righting when tipped to sides 224 Supine: rolls to prone 225 Prone: pivots 226 Prone: crawls 227 Sitting: protective extension to the sides 228 Standing: moves body up and down 229 Sits unsupported for 60 seconds 230 Prone or sitting: assumes quadruped position 23I Quadruped: equilibrium reactions 232 Sitting: assists in pulling upright 233 Prone or quadruped: assumes sitt'mg position 234 Standing: raises one foot (attempts to step) 9-II Merrill 235 andl'tlped: creeps 236 Sitting: protective extension to the rear 237 Sits alone and steady I0 minutes 238 Sitting: pulls to standing using furniture 239 Standing: lowers self to floor 240 Standing: cruises by holding onto funitwe ”I Wake With one hand held 242 Sitting: equilibrium reactions 243 Stands alone [arm-d Yam“!- W thmwm 106 I. Maser um: WALLEVEIsANDm 3‘" 9‘“ 0'“ D“ I2-I5 Months 244 Walks by himself 245 Creeps up stairs 246 Standing: throws ball with some cast 247 Walks well (stops. starts. turns) 248 Supine: raises self to standing position independently 249 Walks backward I6-I9 Months 250 “Runs" stiffly 25I Walks sideways 252 Walks up stairs held by one hand 253 Creeps backward down stairs 254 Standing: seats self in small chair 255 Climbs into adultqize chair 256 Standing: balances on one foot with help 257 Standing: equilibrium reactions 20-23 Mondis Lzsr Walksdownstairswithonehandheld I259 Squats in play: renames standing position Jumps in place 24-27 Months [Tet Goesupanddownstairsalonenonreciproally r 262 Stands on balance beam with both feet; attempts to step Kicksball W“ Jumps from bottom step (both feet together) 28-31 Months F265 Walks on tiptoes ThrowsballSto‘lfeatinsvertieslpatrem Whlwndfmafln 'hl’WWWb-flr 107 Or. Maser "m DEVELOPMENTAL LEVELS AND U’EMS 0‘“ D‘" 0‘“ D‘“ 267 Takes a few alternate steps on balance beam 268 Supine: rises to standing with mature pattern 32-35 Months 269 Rides tricycle using pedals 270 Goes up stairs alternating feet 27l Stands on one foot and balances 272 Walks with heel-toe gait 273 Walks with redprocal arm swing 274 Runs hop-rm for Infants and Young Mn» Vane 2; Edy Ines-narro- Derebpmral ho/ilr 108 Profile Graph Nun mom Munster 33' m1. WW Wm m 531., ram. $2327.: um... 52?: ao- l07 to: I90 274 32-35 73 I I m I I so I03 no l88 269 12 l02 268 28-3l as I” no m m I 7! not ass :7 to too l38 m 264 24.27 I I too its I so or so m m 261 as or US I85 zoo 10.23 I I rs I68 I75 I so u 132 I83 zsr :9 s3 94 m 251 l6-I9 I I I m I m m as as I65 2:0 as s7 l30 m m m 2.9 1 l l I l t 2l st to 129 l62 m m 244 so :1 r: us lbl 243 t t I l 1 l6 s: at us I” as l5 5l so 124 m 234 .. i l t t i II as 19 m m 2)!) to 45 is no us our ’4 I I 1 I 2 4: rs "2 I43 m m m or I or 14 nor l9l Numbers Whlnfinadfmafin WW V‘- 2. ‘00 in!“ 109 Appendix A.4 Parent Survey # 2 3 Months PARENT SURVEY EMILY CME MEN “TE 2 THE RINSE OF THIS WESTIOIOIAIRE IS TO IDENTIFY THE EFFECTS THE BIRTH OF AN INFANT HITH SPINA IIFIOA IN THE RE OF THEIR CHILD. NIELWYSPLASIA. PART I MYELODYSPLASIA) HAS ON A FAMILY AND TO IDENTIFY HHAT FHILIES EXPECT OF PROFESSIONALS YOUR COMPLETION OF THE QUESTIONNAIRE HAY GIVE US SOME SUGGESTIONS ”OUT PROVIDING THE TYPE OF ASSISTAKE THAT MAY BE HELPFUL TO YOU AS A man or A CHILD HITH TIE FIRST SERIES 3 WESTIONS ASKS 504E 8ACKGROUNO WESTIONS ABM rou. PLEASE CHECK THE APMPRIATE RESPONSE 0R FILL IN THE EXACT NUI‘BERS AS INDICATED. I. 4. that Is your relationship to the child with nyelodysplasia? Ihther _ Father Diet is the year of my; birth? Give exact tuners. year list category mst closely describes pur decimation? (Check only ON__§)(I3) Uhsktllad worker (l) Seal-skilled worker (2) Skilled worker (3) __Professional (4) _Grner of business (5) ___Ilort in business (spectfyHG) __Other (speciryIrgI (a) be you anoyed row? (1a) ___I'0 (2) If yes: __Full arts” ne __Othar (specify) hat is the hlwest educational level you have mleted? (Cheek only fit) ()6) __8th grade or less (I) —9-ll grad e (2) __ltt school(3) :1- years post high school“) —Iechelor' s degree (5) _Oegnee(s) beyond Bachelors (6) __Other (specifyll?) MIodysplasta Study I.S.ll. College of Nursing Children's Hospital of Michigan sunossz-ot 7. IO. II. I2. llhat is your present narltal status? (Check only _ON_E) (I7 I lirrled to are father/mother of child with riyelodysplasi‘a (l) _Narrted but not to father/mother of _of child with nyelodysplasia (Z) __Stngle (3) :Slngle but living with father] __Irther of child with nyelodysplasia“) __otvorced (S) :Separatad (6) Ihat is the total W)“ of re ancles yOu or your spouse have had? (lg-19! Exact Mar list is tie total nuber of living children you have now? (20.2” ExactMer Now any children do you have living at ha at the present tine? (22-23) Exact Mar Are there persons other than your spouse and children living with you in your hue? (24) _res (1 Ex? Mer_ (ZS-26) If res. state their relationship to you. (check as nsny as necessary) Your relatives (27) Spouse' s relatives 28) Non-related adultIs) (over )8 years of me) (29 Dn-ralated chlld(res) (under 18 years of age) (1)) I). ll. IS. IS. IT. II. .for each ,itu circle the nine:- 1ndicating how you rate your hue enviroment now. Low High how (space) l 2 3 4 5 ( Neat/cooling l 2 3 4 5 ( Food l 2 3 4 5 Safety 1 2 3 a 5 ( Distance to stores 1 2 3 4 5 ( Clothing l 2 3 4 S ( Now my of your adult relatives (children. parents. aunts. uncles. first cousins. sisters. brothers. grandparents) live within 50 miles of your home? (Check only one) (39) lo relatives (l) l-9 relatives (2) l0 or are (3) lbn't no: how many (4) Have you loved in the sonths since the birth of the child with qyelodysplasia? (Check only one) (so) ....... '05 I Have you or your spouse been in the Nspital in the months since the birth of the child with welodysplasia? (Check only one) (ll) Have any of your other children been in the hospital in the nonths since the birth of the child with myelodysplasia? (Check only one) (42) Yes No: Have any relatives (other than your children or spouse) and/or close friends been in the hospital in the months since 110 lit applicable (no other children)(8) the birth of the child with Iyelodysplasia? (Check only one) (43) ___m (l; b 2 I9. Have you experienced the death of a close friend or close relative in the mnths since the birth of your child with mlodysplasia? (Check only one) (“I Yes (l) lo (2) Since the birth of your child. when yOu spend tin! away from your inediate fanily (spouse and children). was this tin s t with (Check only one last tun . (45) Other family and/or friends (l) Hort and/or school associates (2) Alone (3) Do not spend tine away from (4) i-ediate family (5) How would you rate your physical health now? (Check only one) (46 Poor 1) Fair 2) (3) Excellent (4) How would you rate your erotional health now? (Check only one) (47) Poor :1; Fair 2 Good (3) Excellent (4) I'll PART II THE FOLLONINS QUESTIONS ARE RELATED TO YOUR CHILD HITN MYELODYSPLASIA. I. Hhat is the situation of your child 4. with Ipelodysplasia? (49) cared for in our home (l) and for in nursing hane (or state home) (2) cared for in own home and nursing home (3) cared for in other relative's home (4) cared for in foster home (5) cared for b adoptive home (6) deceased (7 other (specify)(8) To take your child to appoinonents and/or to visit your child do you (Check the 22$:nost common) (50) drive the family car (1) your spouse drives the family car (2) ride with friends and/or relatives (3) ride city bus (4) ride taxi (S) ride special medical transport van (6) do not visit or take to appointments(7) The hospital and doctor costs for an infant with a birth defect can be mrrisclle. costs having on your finances? (SI) It is having no effect as all costs are covered It has a little effect as most costs are covered It has a large effect as many costs are not covered (3 Other (please specify)(8)____________ 5 what effect are these 6 (II)Special care (chde. exercises. etc.) Of the following services (persons). check those that you remember talking to and/or seeing about your child. hospital nurse (53) obstetrician (54) pediatrician (55) public health nurse (visiting nurse)56§ urologist doctor (kidneyssbladder)(57) orthopedic doctor (legs and bones)(58) neurosurgeon (back and head)(59) clinic nurse (60) early intervention school program (61) physical therapist (62) ______occupational therapist (63) social worker (64) orthotist (brace man) (65) religious advisor (pastor. priest. religious counselor) (66) __ speech therapist (67) opthamologist (eye doctor) (68) parent from parent group (69) none(70) Other (specify)(7l) Give the number of the above peo le who came to your home at least once 74-75) lbn't know How would you rate the time you spend on the following activities concerning your child with myelodysplasia. Circle the number for each activity. None Little 0 l 2 I c n 3' 1 don't know the effect (4) I23Playing with l3 Caring for (feeding, bathing. etc.) l4)Horrying about I5)Enjoying (l6)Taking to appointments l7)Talking with your d—D—J-l __lu-l CO 0°00 00 NN NNNN NM spouse E18)Talking with relatives 19)Trying to get babysitters dd Uh) “UN“ Uh! U #5 héb‘ .5 JD aim wmmm U‘m ‘0 A. 112 PART III roe THE FDLLWING QUESTIMS. PLEASE C IRELE TIE MEI! ON THE SCALE IiICN REPRESENTS NOJ TN FEEL m ABOUT THE QUESTIONS film. PLEASE TRY TO ANSHER ALL SCALES. The uount of talk with your friends and/or relatives regarding your concerns and problems. , a. How mch is there now? Little 1 2 3 4 5 Nanchshould there be? Little 1 2 3 4 5 6 7 much (Zl) c. Howinortant is this tone? Little1234567 The uount of time you spend with your use. Hunchis there now? Little L2 3 4 5 6 7 ouch (23) Now loch should there be? Little 1 2 3 4 s 6 7 Inch (24) Now inortant is this to a? Little 1 2 3 4 s a 7 ouch (25) The nomt of discussion of your concerns and problems with your spouse. Nanchistherenow? Little1234567mch Humch should there be? Little1234567luch How inortant is this to me? Little1234567 6 7 ouch (20) such (22) " (as) I’ (27) C. ouch (28) The awount of tile you spend with ”impairs. How-echistherenow? Little1234567ouch liar-achshouldtherebe? Little 1 2 3 4 5 6 Huinontant is this tome? Little1234567 (29) 7 ouch (1)) ‘° Inch (3‘) The mount of time you spend in leisure/ recreational activities. Nowmchistherenow? Little 1 2 3 4 5 6 7 moh(32) Now mach should there be? Little 1 2 3 4 s 6 7 ouch (33) How inortant is this to lie? Little 1 2 3 4 5 6 7 mch(34) l. b. C. The amount of help from your s ouse with fnily tasks such as care of children, house repairs. household chores. etc. How-lien is there now? Little 1 2 3 4 5 6 7 Iuch(35) How much should there be? Little 1 z 3 4 s s 7 luch (36) Now inportant is this to we? Little 1 2 3 4 s a 7 Iuch (37) O. b. C. The amount of help frul relatives (do not include spouse) with Tamil y tasks such as care of children. house repairs. household chores. etc. Hownch is there now? Little 1 z 3 4 s s 7 Iuch (33) How-achshould there be? Little 1 2 3 4 5 6 7 mch(39) liow inortant is this to me? Little 1 2 3 4 s e 7 Inch (40) The anunt of time with health professionals tors. nurses. social workers. etc.) related to your child with wyelodysplasia. a. iiowwchisthere now? Little 1 2 3 4 5 6 7 ouch (43) How ouch should there be? Little 1 2 3 4 s s 7 ouch (4‘) Howth is this to me? Littlel234567 c. m (‘5) IO. II. 12. 13. IA. The haunt of help from your friends with fami 1‘ tasks such as care 0 children. ouse repairs. household Chores. etc. a. How tech is there now Little 1 2 3 4 S 6 7 wch (46) b. Now-loch should there be? Little123456 c. How iloortant is this to he? Little 12 3 4 5 6 7 mch(4a) If you don't have other children. check here 7 wch (a7) l5, l6. and omit questions. ll.lZ.l3. 1. l4. The amount of problems with your g__ther _c_h__ildren. a. How ouch is there now? a Little 1 2 3 5 6 7 mch (49) b. How much should there be? Little 1 2 3 4 5 6 7 mch (so) c. How inortant is this to Ie? Little1234567 The aunt of ti. you spend with your other children. a. flu wch is there now Little 1 2 3 4 5 6 7 mch (52) b. Munich should there be? Little 1 2 3 4 5 6 7 mch(53) c. How inortant is this to me? Little1234567 mch (51) Inch (54) If none of your children are in school. check here and omit question 14. The nount of tile your other children Iiss school. a. Nu mch is therenow Little 1 2 3 5? 6 7 Ilch (55) b. hunch should there be? Little123456 c. liu inortant is this to he? Little1234567 7 NC“ (56) mch (57) I7. I9. 11J3 The aeount of disagree-ants with your SMSO. Lila-l istherenu Little 4 chL23 How-ochshould there be? Little 1 2 3 4 s 6 7 nuchz) c. How ieportant is this to Little 1 2 3 4 5 6 7 mch(13) The aunt of time y_ ouare ill. a. Humuch is therenow Little 1 2 3 4 5? 6 7 euch(l4) b. How Ilcl'l should there be? Little 1 2 3 4 s 6 7 luch(l5) c. How iloortant is this to he? Little1234567 S7 6 7 mch(ll) euch(l6) The anount of time you spend doing house- work (cooking. cleaning. washing. yard- work. etc.). a. hitch is therenu Little 1 .2 3 4 5 6 7 eucn(l7) b. Munich should there be? Little 1 2 3 4 5 6 1 Iach(la) c. Nu inortant is this to la? Little L1 3 4 5 6 7 Theuount of time (including housework a. iiu Inch is there now Little 1 2 3 4 5 6 7 mchiZO) b. Now-ichshould there be? Little 1 2 3 4 5 6 7 mch(21) c. iiu inortant is this to we? Little 1 2 3 4 5 6 7 mch(22) mch(l9) ou mi ss work The awount of tine your s use nisses work. (including housewor a. Munich srethe nu? Little 1‘2 3 4 5 6 7 nich(23) b. Hunchshould therebe? Little 1 2 3 4 5 6 7 lich(24) c. How inortant is this to n? Little1234567-Ich(25) II. The mount of emtional support frai friends and/or relatives. a. How Inch is there now? Little L3 3 4 5 6 7 mch(26) b. Nu lich should there be? Little L2 3 4 5 6 7 ouch (27) c. How inortant is this to oe? Little1234567 The amount of mtional support fru M EON—5' a. How inch is there now? Little L2 3 4 5 6 7 men (29) b. Humch should there be? Little 12 3 4 S 6 7 mchfll) c. Hu imortant is this to ne? Little1234567 The mount of satisfaction with your Irriage. a. How Iach is there now? Little 1 2 3 4 s 6 7 Iuch(32) “all“ should there be? Little 1 2 3 4 5 6 7 such (33) Nuiwortant is this to he? Little1234567 mch (28) wch (3l) ouch (34) 23. 24. 25. 114 The mount of satisfaction with the sexual relations with your spouse. a. How Itch is there now? Little 1 2 3 4 5 6 7 Inch (35) b. How-sch should there be? Little 12 3 4 5 6 7 Iuch(36) c. How inortant is this to he? Little 1 2 3 4 5 6 7 mch (37) boat is mst difficult for you now? (38) lihat is mst helpful for you? (39) THANK YW FOR YWR TIT! IN CWLETIM THIS QUESTIWNAIRE. IF rou HAVE ANY QUESTIUiS OR CUMNTS. PLEASE FEEL FREE TO ASK THE?! OF THE INTERVIEHER C URITE THE! IN THE FQLWING SPACE. Myelodysplasia Study N.S.U. College of Nursing Children's Hospital of iiichigan Ill-i (”632.01 ”77-” 115 Appendix A.5 Parent Survey 3 Months PART I Ti! Pill _ . Hhat category most closely described 7. your occupation? (Check only ONE) (13) _ Unskilled worker (l) Sail-skilled worker (2) Skilled worker (3) Professional (4) __(hrner of business (5) Ibrked in business (specify) (6) Home; and Tim (7) _ooier (specify (a) a. TO THE BIRTH OF THE CHILD HITH MYELODYSPLASIA. d 2. Here you eaoloyed prior to the birth of your child with myelodysplasia? (l4) Yes (l) Ii (2) 3. If yes: (l5) Full time (1) Part time (2) Other (specify) (3) 9. at applicable 4. iliat was the hi¢iest educational level you had coaleted? (Check only _QLE) (16) 3th grade or less (1) 9-ll grade (2) l0. High school (3) l-3 years post high school (4) Bachelor's degree (5) Dawn“) beyond Bachelors (6) 5. Here there persons other than your spouse and children living with you in II your hue? (24) ' Yes (l) he (2) h—ec’t iii-oer (ZS-26) A. If yes. state their relationship to you. (Check as any as necessary) Your relatives (27) Spouse's relatives (28) __ bn-related adult(s) (over la a? of age) (29) related child(ren) (wider l8 3mm of use) (30) Form A FAHILY cult men on: FATHER __ ionicn _ m A ORHEXT SEVERAL WESTIONS ARE RELATED TO YDLR ACTIVITIES AND CMCERNS IN THE YEAR (I2 m5) TRY TO RECALL AS BEST Y0.) CAN. For each item circle the nuwer indicating hu you rated your Eda enviromnt. Lu High kid-(space) l 2 3 4 6 33 Heat/cooling l 2 3 4 5 34 Food l 2 3 4 S 35) Safety l 2 3 4 5 36) Oistancetostores l 2 3 4 S 37) Clothing l 2 3 4 6 38 Now any of your adult relatives (children. parents. aunts. uncles. first cousins, sisters. brothers. grandparents) lived within 50 miles of your hue? (39) lo relatives (l) l-9 relatives (2) lo or more (3) ain't know how many (4) Have you saved in the year (12 months) prior to the birui of the child with myelodysplasia? (Check only ONE) (40) Yes (l) — ho ( 2) Have you or your spouse been in the hospital in the year (12 months) prior to the birth of the child with myelodysplasia? (41) Yes (l) __ib (2) Have any of your children been in the hospital in the year (I! nonths) prior to fleabirth of the child with myelodysplasia? not a liable (no other children)(8) Yes (l _' I0 (2) Mlodysplasia Study H.S.U. College of Nursing Children's Hospital of Hichigan lllil 00632-0l I2. 13. I4. LISTED aELou ARE sore QLESTIOiS RELATED TO THE mi: serous THE BIRTH or voun CHILD. mm 115 Have any relatives (other than your children or spouse) anger close friends been in the hospital in the yearTle anths) prior to the birth of the child with myelodysplasia? (43) Yes (1) _IO (2) Did you experience the death of a close friend or close relative in vie year prior to the birth of your child with myelodysplasia? (44) Yes (i) No (2) Prior to the birth of your child. when you spent time away from your inmediate faily (spouse and children). was this tia spent with (Check HOS__T canon) (45) _Other family/or friends (l) :Hork/or school associates (2) —Alone (3) _Oid not spend time away from —iaediate family (4) l5. 16a PART II mm: mandala: soul: wilcii nerncsms now LLEASE TRY TO Ansutn ALL scams. 1. The mount of talk with your friends and/or ___W latives regarding your concern a %5Iems. a. (20) Howmuch was there? Littlel234567much b. (2l) How mach should there have been? LittlelZ34S67much c. (22) How isoortant was this to we? Littlel234567much The aaunt of tia you spent with your 1293—.- a. (23) How much was there? Littlel234667much b. (24) How much should there have been? Littlel23 567mch c. (25) How iaortant was this to me? Little L2 3 4 5 6 7 much 3. Before your child was born. how would yourate your )phfiical health? (Check only __) (46) I) Fair 2) Good 3 Excellent (4) Before your child was born. how woold your emotional health? (Check only _iig) (47) Poorl Fair 2 Coed 3 Excellent (4) PLEASE EIIAS RATED. The amount of discussion of your concerns and problas with your gpo_use_. a. (26) How much was there? Littlel234567much b. (27) How mach should there have been? Little 1 2 3 5 6 7 mach c. (28) How important was this to a? Littlel234567wch The aunt of time spent with gighbors. a.(29)liumuchwasthere? Littlel234667muoh b. (m) Hu much should there have been? LittIeI234567much c. (3l) iiu iaortant was this to me? LittleL234567much 7. The amaunt of time you spent in leisure/ recreational activities. a.(32)Howmuch was there? Littlel234567much b. (33) How mach should there have been? LittleI234567liuch c. (34) How important was this to me? Littlel234567mach The alount of help from your s ouse with family tasks such as care of children. noise repairs. household chores. etc. a. (35) How much was there? Littlel234567mach b. (36) How much should there have been? a Little 1 2 3 5 6 7 much c. (37) How iloortant was this to me? Little l 2 3 4 5 6 7 much The aaimt of help from relatives (do not include yuar spouse) with family tasks such as care of children. house repairs. household chores. etc. a. (36) Nu mach was there? Little1234567mach b. (39) How mach should there have been? Littlel234567much c. (40) How iaortant was this to me? Littlel234567much 117 ID. ‘1. l2. l3. l4. The aunt of _t___ime with health professionals (doctors. nurses "—social workers. etc.) related to your health problems. a. (43) How mach was there? Littlel234567mach b. (44) How much should there have been? Littlel23 S67Iluch c. (45) How iaortant was this to me? Littlel234567 T.he aamt of help from your friends with faily tasks such as care of children. house repairs. household chores. etc. a.(46)iiowmuchwasth Little1234?567much b. (47) How mach should there have been? Little123 567much c. (40) How iaortant was this to re? Little 234567-4ch mlch l5. I6. If you don't have other children. check here and unit ll. l2. l3. and 14. The alount of problas with your other children. a. (49) How much was there? Little1234567much b. (50) How much should there have been? Littlel234567naach c. (Sl) How iaortant was this to me? Little1234567 The aaunt of time you sent twith your other children. a. (52) How much was there? Littlel234567much b. (53) How much should there have been? Littlel234567much c. (54) How imortant was this to me? LittIeI234567 much much if none of your children are in school. check here and omit question 14. The anount of time your other children missed school? ""—"' a. (55) How mach was there? Littlel234567lluch b. (56) How mach should there have been? Little1234567much c. (57) How iamortant was this to me? Little1234567much The amaunt of disagreaents with your Mo a. (ll) How mach was there? Littlel234567much b. (12) How mach should there have been? Little 1 2 3 4 5 6 7 much c. (l3) How iaortant was this to me? Little1234567 The aunt of time mare ill. a.(l4)iiowmachwasthere ? Little1234567much b. (l5) Iiu much should there have been? Littlel234567much c. (l6) Hu iaortant was this to me? Little1234567much mach 17. IO. ‘9. The hunt of time you spent doing housework (cooking. cleaning. washing. yaruork. etc.) a. (I?) Humach was there? Littlel234567much b. (It) How much should there have been? Littlel234567much c. (I9) lbw impartant was this to me? Little1234567much The mount of time lag missed work (including housework a. (20) How mach was there? LittIeI234567aach b. (21) How mach should there have been? Littlel234567much c. (22) Now ieportant was this to me? Littlel234567mach The aoimt of tire yoor s use missed work (including housework a. (23) flu mach was there? Littlel234567much b. (24) How much should there have been? Little1234567much c. (25) lbw inortant was this to me? Little1234567mach The aount of emotional support from fring and/or relatives. a. (26) How mach was there? Little1234567mach b. (27) How much should there have been? L1ttIeI234567much c. (28) How imiortant was this to me? Littlel234567mach 118 2l. 22. 23. THE MWIIB NESTIDNS RELATE TO YOU! PREGNANCY. I. Ilat was ur general feeling about the pregnancy and the baby 38) _ _ ___yEas pregnanc _be all right Easy pregnancy but felt there was suething wrong with baby (2) Difficult pregnancy but thOught the baby would be all right (3) was meaning wrong with baby (4) Don't knu (5) 30d thought baby would Difficult pregnancy and thought there The aaunt of eational support from your sause. a. (29) Humachwas there? Littlel234567much b. (3)) how much should there have been? Little1234567mach c. (31) How imaortant was this to me? Little1234567 The amount of satisfaction with your arriage. a. (32) How mach was there? Little1234567much b. (33) Now mach should ti‘lere have been? mach Little I 2 3 5 6 7 much c. (34) How important was this to me? Little L 2 3 4 S 6 7 mach The amiunt of satisfaction with the sexual relations with your spouse? a. (35) Nu mach was there? Little1234567much b. (36) How mich should there have been? LittIeI234567much c. (37) How important was this to me? Little L2 3 4 5 6 7 mach what was mg smse' s general feeling about the pregnancy and the baby. (39) Eas pregnancy and thought baby wou d be all right (1) Easy pregnancy but felt there was scathing was wrong with baby (2) Difficult pregnancy but thought baby would be all right (3) Difficult pregnancy and thought there was something wrong with baby (4) __mn' t knu (5) 119 5 3. The doctor and hospital costs for a 4. Hhich pregnancy resulted in the child with sreggancy can be worrisome. Hhat effect welodysplasia? (41-42) these costs have on your finances? (Check only 95;) (40) ‘ Exact number ' It had no effect as all costs were 5. Has the pregnancy which resulted in the covered by insurance or Medicaid (1) child with myelodysplasia a planned it had no effect as we planned for pregnancy? (43) the costs (2) Y s (I) It had some effect. as same costs — I: (2) were not covered (3) —— — 2:30: mfiéffigt' " “W “m a. what is the birth date of your child ain't know (5) with myelodysplasia? n W (44-46) (46-47) PART 111 THE NEXT SEVERAL QUESTIONS ARE RELATED TO YOUR ExPERlENCES AT THE TIME YOUR CHILD HAS BORN. l. iihen were you told your child had a 4. iihat is the distance you traveled from birth defect? (51) your hone to Children's Hospital? (Dr to W09 Pm“! “m", nonth) (1) other hospital if not transferred) (63) __ Less than 50 miles (I) Duringjabor (Z) 51 to 100 miles (2) Delivery room (3) Over 1w miles (3) —"within first 24 hours (4) ‘— 2-3 days (5) S. Ilat were you told regarding having After 3 days (specify time) (6) contact with your child while she/he was in the hospital? (64) 2. Hho told you your child had a birth _S“ ”4 care for child as M" ” ”NE? ”A. “CITE ( ) _See and care for the child a few Doctor who delivered infant (56) times (2) Pediatrician (57) See and care for the child once (3) Neurosurgeon (58) Do not see and care for the child —nurse (59) —at UT (4) Spouse (60) _PIM" (Specify) (51) ' 6. Hhat were you told to expect regarding your child? (65) hfect can be surgically closed (1) 3. How old was your child when transferred _— 0“.“ can be surgically closed but to Children s Hospital? (62) suroerv not rec nded I2) Hithin first 24 hours (1) Defect cannot be surgica 1y closed 2 days (2) but child will be re-evaluated (3) _ 3 days (3) __ Defect cannot be surgicall closed 4 day under any circlmlstances (4) _ s (4) _ Other (Specify) (5) ufect cannot be sur ically closed. at transferred to Children's Hospital no check-ups needed 120 7. Hhat are you told were the possibilities 12. If yes. how old was your child when you for care for your child? (Check _a_ll learned about the opportunity to talk possibilities you were told) with other parents? (12) Place the child in a nursing hone (66) Iithin first 14 days after birth (1) Place the child in a State Hospital 15 days to 1 month (2) or institution (67) __5 weeks to 3 months (3) Take the child hone (68) :Over 3 anths (4) Child would not leave the hospital (69) :lbn t rememier (5 ) Place the child in foster care (70) Place the child for adoption (71) 13. if you were informed about other parents. Other (specify) (72) idi__g_ told you? (Check as any as necessary) m informed (131)” 8. Hhat was most hel ful to you the first -—- ther ”'9'“ few weeksmild was born? (73) __Social “'1‘" (‘5) —Ooctor (l6) _hln-se (l7) :Friend (18) :Spouse (19) _Other family alter (20) hws media: TV. Radio. Newspaper (21) 9. iihat was least hel ful to you the first few weeks after the child was born? (74) 14. iihen did you first talk with another parent of a child with myelodysplasia? (22) __Hithin the first week (1) _Two weeks to one month (2) Hithin the first six months (3) After first six nonths (4) ID. Hu would you describe your child' so“ Have not talked with another parent present condition in terms of howy (5) are told it would be? (Check only yp__NE) (75) Here you given written information about l.5 —::::ii a: it: :1.‘?.L2:2312.2:°?§?”’ .....,....... W m" <2” —Child alive and better than told (3) Yes (1) __Child alive and sea as told (4) No (2) —Child alive and worse than told (5) Don‘t knu (3) __Child alive with different problems Other (specify) (4) —than told (6) II. Here you informed of the opportunity to l6 He . re you infomed of the possibility of & 31:5"??1?‘ children "It" a" financial assistance for the special ' adical expenses with your child? (24) —.'.:‘ i" _v-s m i ) __Don't reader (3) —:nI:)tm (3) I. 4. 12‘) PART IV LISTED DELW ARE SO! QUESTIOIG RELATED TO YOUR INVOLVEKNT HITH YOUR CHILD AND HEALTH PRNESSIONALS AT THE TIME YOUR OiILD HAS BORN AND THE FIRST FEli DAYS FOLLOJING THE BIRTH. :LEIHEE CIRCLE THE NUI‘BER (Ii THE SCALE WICH REPRESENTS HON YOU FEEL ABOUT THE QUESTIONS LLEASE TRY TO ANSHER ALL SCALES. Theneedyouhadtocare foryour chilaa tthe tia of birth. a. (27) How mach was there? Little1234567mach b. (28) How mach should there have been? Littlel234567mach c. (29) How imaortant was this to me? Little1234567luch The amaunt of time you _s_a1 your child. a. (30) How mach was there? Littlel234567mach b. (31) Nu mach should there have been? LittIeIZ34567llach c. (32) Now iaortant was this to a? Little1234567mach aount of tia you t_ou___ched or h___eld your child. a.(33)1iowmuchwasthere LittlelZ34S67mach b. (34) Now mach should there have been? Little1234567much c. (35) How imam-tent was this to me? Littlel234567mach Encouragement from the do_c___tors u see your child. a. (36) Now mach was there? Littlel234567mach b. (37) How mach should there have been? Littlel234567IOch c. (34) How imaortant was this to me? Littlel234567mach EncOuragament from the n__urses to see your child. a. (39) How mach was there? Little L2 3 4 5 6 7 mach b. (40) Nu mach should there have been? Little I 2 3 4 5 6 7 much c. (41) How iaortant as this to me? Little I Z 3 4 S 6 7 much Ehnfigragaent from spguse to see your a. (42) How mx:h was there? Littlel234567muCh b. (43) How mach should there have been? Littlel234567mach c. (44) Hu inortant was this in me? Little1234567mach Encouragement from famileembers/friends (other than spouse) to see your child. a. (45) how mach was there? Littlel234567mach b. (46) How mach should there have been? LittIe1234S67much c. (47) How iaortant was this to me? Little1234567much The opportunity for you as a parent to decide if the child was to have surgery. a. (48) liu much was there? Littlel234567much b. (49) How mach should there have been? Little1234567mach c. (50) Now iaortant was this to a? LittleL234567mach 122 9. The opportimity to decide abut placement for your diild (i.e., home. hospital. nursing hole. institution). a. (51) Now mach was there? LittleL234567mach b. (62) How mach should there have been? Littlel234567mach c. (53) How iaortant was this to me? Littlel234567mach THE HEAT FEH WESTIOIS ARE RELATED TO THE TIME A FEH HEEKS AFTER THE BIRTH OF YOUR CHILD. l. The aunt of encouragement to care for 3. During the first few weeks aft—e__r your your child a few weeks after birth. child was born. hu would you rate a. (5‘) How much was there? your physical health? (Check only ONE) (62) Littlel234567mach Poor(l) b. (57) liow mx:h should there have been? — _2;; g c. (S!) ikaw iaortant was this to a? Little 1 2 3 4 5 6 7 mach 4. During the first few weeks e_fter your child was born. how would yo—u ra 2. The need you had to care for your child ymar aotional health? (Check only 9__) (63) a few weeks after birth. a. (59) Now mach was there? Fair (2) Littlel234567mach b. (60) how mach siould there have been? Littlel234567mach c. (ET) How important was this to a? LittI¢I234567lxii Good (3) Excellent (4) m YOU FOR YOUR TIIE IN CMETIN THIS WESTIONNAIRE. IF rou I‘VE MY WESTIONS OR CWENTS. PLEASE FEEL FREE TO ASK THE! OF THE INTERVIEHER on NRITE THEN IN THE FDLLGIING SPACE. melodywlasia Study N.S.U. College of Nursing Children's Hospital of Hichigan NIH ”32-01 ION-Q PROFESSIONALS IN THE CARE OF THEIR CHILD. 123 Appendix A.6 Parent Survey # 3, 4, 5 6, 12, IB Months PARENT SURVEY THE PURPOSE OF THIS QUESTIONNAIRE IS TO IDENTIFY THE EFFECTS THE BIRTH OF AN INFANT HITH SPINA BIFIDA (MYELODYSPLASIA) HAS ON A FAMILY AND TO IDENTIFY HHAT FAMILIES EXPECT OF FANILY coo: men m:n_mnzn_ pm 3 4 5 YOUR COMPLETION OF THE QUESTIONNAIRE MAY GIVE "5 SOME SUGGESTIONS ABOUT PROVIDING THE TYPE OF ASSISTANCE THAT MAY BE HELPFUL TO YOU AS A PARENT OF A CHILD HITH MYELODYSPLASIA. PART I THIS SERIES OF QUESTIONS ASKS SM BACKGROUND QUESTICNiS ABOUT YOU. PLEASE CHECK THE APPROPRIATE RESPONSE OR FILL IN THE EXACT NUMBERS AS INDICATED. I. 2. J4. Ihat category most closely describes your occupation? (Check only QN§)(13) Unskilled worker (I) Semi-skilled worker (2) Skilled worker (3) fessional (4) r of business (5) —Ilork in business (specify) (6) Home and famil (7) r (specify (8) Are you moloyed now? (I4) Yes I —.. (2) If yes: Full time (1) (15) art time (2) —Dther (specify) (3) Nhat is the highest educational level you have mleted? (Check only ONE) (16) 6th grade or less (1) 9-11 grade (2) -_|:iigh school(3) i 1 ( ) - years post h gh schoo 4 bachelor's degree (5) —bgree(s) beyond Bachelor's (6) other(specify)(7) M10475” asia Study N.S.U. College of Nursing Children's Hospital of Hichigan NIH 00632-01 lihat is your present marital status? (Check only gym) Narried to the father/mother of child with myelodysplasia (1) Married but not to father/mother of child with myelodysplasia (2) Single (3) Single and living with father/mother of child with myelodysplasia (4) Divorced (5) rated (6) "_bther (specify) (7) Hhat is the total number of re nancies you or your spouse have had? (la-IS) Exact nimiber Nhat is the total number of living children you have now? (20-21) ‘ Exact nimber How many children do you have living at home at the present tine? (22-23) Exact nulber Are there persons other than your spouse and children living with you in your home? Yes (1) (2‘) 2 5. act iii-liar)- (ZS-26) IO. THE EXT WESTIONS HILL BE RELATED TO YOUR ACTIVITIES AND COMERNS SIICE YOU WPLETE LAST WESTIOMAIRE (3 or 6 KINTHS). l2. ‘3. IS. IS. If yes. state their relationship to you. (Check as many as necessary) Your relatives (27) Spouse's relatives (28) of age) 29) Ian-re ated child(ren) (under 18 years of age) (30) Now many of your adult relatives (children. parents. aunts. uncles. first cousins. sisters. brothers. grandparents) live within 50 miles of your hoa? (Check only Q_N_E_) (39) No relatives (l) 1-9 relatives (2) 10 or are (3) M't knu how many (4) Have you aved in the last 3-6 months (Check only at). (40) z: I? (I) Have you or your spouse been in the hospital in the last 3-6 months (Check only 91;). (41) — r: u Have any of your other children been in the hospital isn) the last 3-6 manths (Check only fl). :2’8) Non-related adult(s) (over 18 years 124 ll. 18. ‘9. __ lat applicable (no other children)(8) Have any relatives (other than your chian or spouse) and/or close friends been in the hospital in the ast - anths (Check only .ON_E_). (43) :1? (I) For each item. circle the hunter indicating how you rate your hone envirorment nu. Lu High Room (space) 1 2 3 4 (33 Heat/cooling l 2 3 4 5 (34 Food I 2 3 4 5 (35 Safety 1 2 3 4 5 (36 Distance to stores 1 2 3 4 5 (37 Clothing 1 2 3 4 5 (38 0111: TRY TO RECALL AS BEST YOU CAN. I7. Have you experienced the death of a close friend or close relative in the last 3-6 months (Check only gig). (44) 'Yes 1) Na 2) iihen you spend time away from your imnediate family (spouse and children). is this time spent with (Check most comnon). 5 Other family/or friends (1) Hark/or school associates (2) Alone Do not spend time away from i—diate family (4) Nu would you rate your physical health now (Check only 911; . Poor I) Fair 2) _ (3) Excellent (4) Nu would you rate your errotional health now? (Check only O_N§_). (47) Poor 1) —Fair 2) d 3) Excellent(4) I25 PART II THE FQLOHIB WESTIM ME RELATED TO roux CHILD HITH MYELODYSPLASIA FOR THE TIHE SINCE YOU COMPLETED THE LAST QJES‘IIGINAIRE (3 OR 6 IDNTHS) 4. I. what is the situation of your child with myelodysplasia? (49) cared for in our home (l) cared for in nursing home (or state home) (2) cared for in own home and nursing home (3) cared for in other relative's home (4) cared fur in foster home (5) cared for by) adoptive hale (6) deceased _other (specify) (§) To take your child to appointments and/or to visit your child do you (Check the one most conmon). (50 0) ___drive the fmni 1y car (1) —your spouse drives the family car (2) ride with friends/or relatives (3) ride city bus (4) ride taxi (5) ride special medical transport van (5) do not visit or take to appointments (7) other (please specify) (8) The hospital and doctor costs for an infant with a birth defect can be worrisome. Hhat effect are these costs having on your finances? (51) It is having no effect as all costs are covered It has a little effect but most costs are covered It is having a large effect as many costs are not covered 1 don't know the effect 0f the following services (persons). check those that you renenber talking to and/or seeing about your child since u completed the last questionnaire 3 or 6 months). _hospital nurse (53) :obstetrician (54g :pediatrician 55 _public health nurse (visiting nurse)56) urologist doctor (kidneyshbladder)(57) orthopedic doctor (legs and bones)(58) neurosurgeon (back and head)(59 ) clinic nurse early intervention school program (61) physical therapist (62) occupational therapist (63) social worker (64 ) orthotist (brace man)(65) religious advisor (pastor. priest. religious counselor)(66) _speech therapist (67) _opthalmologist (eye doctor)(68) parent from parent group (69) none (70) other (specify)(7l) Give the number of the above peo 1e who came to your home at least once 74-75) Don't know child with myelodysplasia? Circle the nuifir Tor each activity. None Little Much (11) Special care (crede. l 2 3 4 5 exercises. etc.) (l2; Play im with O 1 2 3 4 5 l3 Caring for (feeding. bathing. etc.) 0 l 2 3 4 5 (l4) worrying about 0 l 2 3 4 5 IS) Enjoying 0 l 2 3 4 5 16) Taking to appointments 0 l 2 3 4 5 (17) Talking with your 0 l 2 3 4 5 l8 Talking with relatives 0 l 2 3 4 5 l9 Trying to get 0 l 2 3 4 5 babysitters 126 PART III For THE FOLLOHIHG outsnons. PLEASE ClRCLE THE nuance on THE SCALE lliICH museum now You FEEL Lay, ABOUT THE ouzsnous sumo. ”E TRY TO ANSHER ALL SCALES. I. 2. The aount of talk with your f_r____iends and/or relatives regarding your concerns a problems. a. How much is there now? Little 12 3 4 5 6 7 mach (20) b. How mach should there be? Little 1 2 3 4 5 6 7 mach (21) 5. Nu iaortant is this to me? Little 1 2 3 4 5 6 7 much (22) The aount of time you spend with your m- a. Nu mach is there nu? Little 1 2 3 4 s 6 7 mach (23) b. Humach should there be? Little 1 2 3 4 5 5 7 mach (24) c. Huiaortant is thistome? Little1234ss7nuchizs) The aount of discussion of your concerns ad problems with your spouse. a. Humachistherenu ? Little L2 3 4 s a 7 mach (26) b. Numach should there be? Little1234567mach c. Nu inortant is this to me? LittleL234567 (27) mach (23) 14. Theaauntoftimeyouspendwith MID—59.9. a. Humachistherenow? Litt141234ss7wuchizsl b. Humach should there be? Little1234ss7wuchiall c. Nuiaortant is this tome? Little 1 2 3 4 SL7 mach (3}) The mount of time you spend in leisure/ recreational activities. a. Humach is therenu Little 1 2 3 4 5 6 7 mach (32) b. How mach should there be? Little123456 c. How iaortant is this to me? Little 12 3 4 5 6 7 mach (34) 7 much (33) The amount of help from your s ouse with faily tasks such as care of children. house repairs. household chores. etc. a. How mach is there now? Little 1 2 3 4 5 b. Hu mach should there be? Little 123 4 5 6 7 mach(36) c. Howth is this to me? Little1234567 The amount of help from relatives (do not include spouse) with family tasks such as care of children. house repairs. household chores. etc. a. Humach is therenu Little 1 2 3 4 5? 6 7 mach (38) b. Numach should there be? Little 1 2 3 4 5 6 7 mach(39) c. Nu iaortant is this to me? Little I 2 3 4 S 6 7 mach(40) The amaunt of time with health professionals Waiters. nurses. social workers. etc.) related to your child with myelodysplasia. a. liowmachis there LittlelM234sa7-iohm) b. Humachsiwauld there be? Littlel234567machi“) c. Nuiaortant is this towe Little1234567machi45) 6 7 mach (35) mach (37) IO. II. 12. 13. I4. The aunt of help frrml your friends with fail‘otasks such as care of children. use repairs. household m. .tCe a. Nu mach is there nu? Little 12 3 4 5 6 7 mach (46) b. Humach should there be? Little123456 c. Nu iaortant is this to me? Little 1 2 3 4 s 6 7 much (48) If you don't have other children. check here—— 7 mach (47) The aount of problems with your p___ther Qildren a. Nu mach is there nu? Little I 2 3 4 5 6 7 mach (49) b. Humuch should there be? Little I 2 3 4 5 6 7 mach(SO) c. Howiaortant is this tome? Little1234567 Themnt of timeyouspend with your otherchid.lren a. Numachistherenu Little I 2 3 4 s 6 7 mach (52) b. Humach should there be? Little 1 2 3 4 5 6 7 mach (53) c. How imaortant is this to me? Little 12 3 4 5 6 7 mach(54) If none of your children are in school. check here and uit question 14. The aount of time your other children miss school. a. Humach Little I 2 3" 4 5 6 7 mach (55) b. Numachshould there be? Little g 3 4 5 6 7 mach (56) c. ibwiaortant is thist me? Little 1 2 3 4 5 6 7 mach(57) mach (51) 127 15_ I6. and omit questions. 11.12.13. s l4. 17. 18. IS. The mount of disagreements with your spggse. How mach is there Little I 2 3 b. Humach should there be? Little123456 c. How iaortant is this to a? Litt1e1234567 The mount of time m are ill. a. How mach is therenu Little 1 2 3 4 5 6 7 mach (14) b. Humach should there be? . Little 1 2 3 4 5 6 7 much (15) c. Nu iaortant is this to we? Little 1 2 3 5 6 7 The mount of time you spend doing house- work (cooking. cleaning. washing. yard- work. etc. ). a. How mach is therenu Little IJ 3 4 5 6 7 mach (17) b. Humachshould there be? Little 1 2 3 4 5 6 7 mach(la) c. How inoortant is this to me? Little1234567 u miss work 4 5? 6 7 mach (ll) 7 mach (12) mach (l3) much (16) mach (19) The amount of tia (including housewor a. How mach is there now Little 1 2 3 4 b. Humach should there be? Little 1 2 3 4 5 6 7 mach(21) c. Nowimaortant is this tome? Litt1e1234567 5 6 7 mach(ZO) mach (22) The aunt of time your s e misses work. (including housewor a. Huma sereth nu? Littled‘ I 2 3 4 5 6 7 mach(23) b. Humachshould there be? Little 1 2 34 5 6 7mach(24) c. Nuiwportant is this tome? Little1234667mach(zs) 21. 128 The aunt of eational support from friends and/or relatives. a. How mach is there nu? Little L3 3 4 5 6 7 mach (26) b. Numachshould there be? Little L2 3 4 5 6 7 mach (27) c. How iaortant is this to me? Little 1 2 3 4 5 6 7 mach (28) The amount of emotional support from ”I" m- a. How mach is there now? Little I 2 3 4 L6 7 mach (29) b. Humach should there be? Little 1 2 3 4 5 6 7 much(3)) c. How inortant is this to me? Little 12 3 4 5 6 7 mach (31) The mount of satisfaction with your arriage. a. How mach is there nu? Little 12 3 4 5 6 7 much (32) b. Humach should there be? Little 1 2 3 4 5 6 7 mach (33) c. Nuiaortant is this to a? Little 1 2 3 4 5 6 7 mach(34) 24. 25. The aount of satisfaction with the sexual relations with your spouse. a. How mach is there now? Little 1 z 3 4 s 6 7 mach (35) b. Humachshould there be? Little123456 c. How iaortant is this to me? Little 1 2 3 4 5 6 7 mach (37) iaiat is ast difficult for you now? (38) 7 mach (36) lliat is ast helpful for you? (39) THANK YOU FM YWR TIIE IN CUPLETING THIS QUESTIUINAIM. IF rou HAVE ANY MSTIMS OR CMNTS. PLEASE FEEL FREE TO ASK THEN OF THE INTERVIEHER on URIT'E THEN N THE REVERSE SIDE. Ryelodysplasia Study N.S.U. College of Nursing Children's Hospital of Hichigan NIH Meal 1377-!) 129 Appendix A.7 Profile 0 Hood States (0145) Neonatal. 3. 6. 12. 18 loathe c - N or m w a -~ 0 0 NAME DATE ‘ ' ‘ ' ‘ " s c - 0. 4' m w _, e e. Below isalisl oi words that describe ieelings people have. Please E, c ' " ' " ‘ ' " read each one carefully. Then fill in ONE space under the answer lo 5 ° " ‘ " ‘ " " S " the righl which besl describes HOW YOU HAVE BEEN FEELING OUR- z . ‘ 7 mo THE PAST weex lNCLUOING TODAY. 6 r: - .. . - O u N ' m .‘ .l :4 a- , :3 " > Thenurnbersroier 345'}? 2.55; lolheeephraaas if‘pg if‘pg o-mm-u 2:525 2:125 l-Aliiile oiasa O‘Eas 2-Moderalaly 21 Hope s . 45. Desperate 3-Oulloabli o i z s a 2 s 4-Exlrernely 22. Relaxed . . 46. Sluggish . 5 '- > > ° ‘ 3 ’ ‘ c i 2 - 4 s - .. U orl . . 47. Flebalhous tugis _ w z 4: g o i 2 s 4 , c l . 2 4 T § : g i g; 24 Spite ul . . . 46. Helpless . . ( lea 0 l 2 3 4 D l 2 3 4 C 2 3 4 l. Friendly 2 Sympathetic . . 49. Weary 0 3 D i 2 3 4 O l 2 3 4 2. Tense \26 Uneasy 50. Bewlldered D l 2 3\4 o l 2 3 4 C l 2 3 4 o 3. Angry . . . \ 27. Restless . . . 51. Alert . . . l 2 4 0 1 2 3 4 0 ‘- 2 3 4 4. Worn ouif- . . / 28. Unableioconcenirale 52. Oeceived. . . D 1‘2 3 4 D l 2 3 4 0 l 2 3 a 5. Unhapp . . .< 29. Faligued . . . 53. Furious . . . l 2 l 4 O i 2 J 4 O 1 2 3 4 A 6. Clear-headed . . 30. Helpiul . . . 54. Efficient . 0 1 2 3 4 o l 7 3 a c i 2 3 4 7. Lively \/ . 31. Annoyed . . 55. Trusting . 0 l 2 3 4 0 l 2 3 4 D l 2 3 4 8. Confused. . . 32. Oiscouraged . . 56 Full of pop T i 2 3 4 0 l 2 3 4 o ‘ 2 3 4 v 9. Sorry for things done 33. Resaniiul. . . 57. Bad-lempered. D i 2 3 4 0 l 2 3 4 o l 2 3 4 10. Shaky 34. Nervous . . . 58. Worthless 1 i 2 1 4 o i 2 3 4 o l 2 1 4 11. Llelless 35. Lonely 59. Forgeiiul. oies4 oizs4 01234 p 12. Peeved . . . 36. Miserable . . 60. Oareiree . D T 2 1 4 0 i 2 I 4 O 2*? 4 13. Considerale . . 37. Muddled . . . 61. Terrified . o l 2 3 4 o l 2 3 4 O l 2 3 a 14. Sad. . . . 36. Cheerful . . . 62 Guilty 6 I 1 I 4 D l 2 3 4 O IT? 4 C 15. Active . . . 39. Biiier . . . 63. Vigorous. D l 2 3 4 o l 2 3 4 o i 2 3 4 16. Onedge . . . 4o. Exhausted . 64. Uncertain aboullhlngs D T7 I 4 Tj 2 I f o l 2 3 a 17. Grouchy . . . 41. Anxious . . . 65. Bushed O l 8 I 4 0 i 2 D 4 18. Blue . . . 42. Readylo iighl . . V ‘ Ii 4 D 1 3 Ti MAKE SURE YOU HAVE lo. Energetic. . . 43. Good natured . . ANSWERED EVERY ITEM. 6 i e a 4 o l a s 4 20. Panicky . . . 44. Gloomy G '0“ ”1 POMS common '5 ion eoucATaowAL Ahab awousTeiAL Tesiiwo SERVICE, saw oieoo. ca. mm m sewn APPENDIX B 130 Description of Study to Precede Consent To Be Read to Participant It is known that the birth of a child with a birth defect affects a family in many different ways. The purpose of this study is to learn from families who have had a child with myelodysplasia (spina bifida) and to learn what has happened as a result of this birth. He expect this informa- tion will help to improve the care given to the families and children with myelodysplasia. This study will gather information as to your activities prior to and since the time your child was born, activities at the time the child was born and your current activities and feelings from birth and for 18 months. It will take from 30 to 40 minutes of your time to complete the questionnaire. Information regarding the physical and developmental status of your child with myelodysplasia will also be recorded. The time intervals to complete the Questionnaire are at the baby's birth. 3 months. 6 months, 1 year, l8 months. Your participation in this study is completely voluntary. The information you provide will be identified by a code number to maintain the confidentiality of your opinions. If you agree to participate. you are free to withdraw your consent and discontinue your participation at any time. _ Questions you have about the study will be answered. Do you have any questions at this time? 131 Informed Consent The study has been explained to me. I understand that if I agree to participate. I will: 1. Complete a brief form identifying my feelings. taking 10 minutes today. and at 3 months. 6 months. l2 months. and 18 months after my child was born; 2. Complete a questionnaire about my activities and feelings prior to and since the birth of my child with myelodysplasia (spina bifida). taking 20-30 minutes at 3 months. 6 months. l2 months. and 18 months after my child was born; and 3. The hospital records for my child with myelodysplasia will be re- viewed for the results of the physical and developmental examina- tions. 1 further understand that: all information is confidential and my identity will not be revealed, I am free to withdraw my consent and to discontinue my participation in the project at any time, and any questions I have about the project will be answered. 0n the basis of the above statements. I agree to participate in this project. participant's signature date APPENDIX C 132 Appendix C ' Study Sample Subjects Time 1 2 3 4 5 ' Neonatal 3 months 6 months 12 months 18 months Parents Mothers 27 24 22 25 16 Fathers 19 16 13 12 8 Infants 33 36 30 29 17 * *Number of subjects at 18 months is lower as study is in progress and’ results indicate number reaching 18 months of age. ' BIBLIOGRAPHY » BIBLIOGRAPHY Aldous, J., Osmond, M., & Hicks, M. Men's work and men's families. In w. R. Burr, R. Hill, F. I. Nye & J. L. Reiss (Eds.), Contemporary Theories About the Family (Vol. I), New York: The Free Press, 1979.. Alter, M. Anencephalus, hydrocephalus and spina bifida. Archives of Neurology, 1962, 1, 57-68. Alter, M. Anencephalic birth in a northern and southern community. American Journal of Diseases in Childhood, 1963, 196, 46-54. Apley, J., Barbour, R. F., & Westmacott. Impact of congenital heart disease on the family: Preliminary report. British Medical Journal, 1967, 103-105. Bates, P., West, T., & Schmerl, R. Mainstreaming: Problems, potentials and perspectives. Minneapolis, Minnesota: National Support Systems, 1977. Bell, N. w. & Vogel, E. P. Toward a framework for functional analysis of family behavior. In N. w. Bell and E. F. Vogel (Eds.),.fl modern introduction to the family, New York: The Free Press, 1968. Bohrnstedt, G. N. A methodological note comparing the questionnaire and the interview. Journal of Psychological Studies, 1967, 1§fll), 29-38. Brockelhurst, G. 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