ADAPTATION OF PATIENTS, SIBLINGS, AND MOTHERS T0 CYSTIC FIBROSIS Dissertation for the Degree of Ph.D. MICHIGAN STATE UNIVERSITY ANDREA FARKAS 19 7 3 ”a.” ‘ a This is to certify that the A thesis entitled ADAPTATION or PATIENTS, SIBLINGS, AND MOTHERS TO CYSTIC FIBROSIS presented by 1 Andrea Farkas has been accepted towards fulfillment of the requirements for __ Bh‘D____degree in .EBJEILCLLQQ)’ Major professor Date 8/3/73 /, /’ ’ 0-7639 "Ar 07,: t6”: ‘ ) 33S fiBAO 0 9 320m {fill—HQ}? ‘fllb ABSTRACT ADAPTATION OF PATIENTS, SIBLINGS, AND MOTHERS TO CYSTIC FIBROSIS BY Andrea Farkas The present study examines the effect of the severity of a child's illness on his psychological functioning and the functioning of other family members. Children with diabetes, mild cystic fibrosis, and cystic fibrosis in the terminal stages formed the three main samples. In ad- dition, the mother of each patient and one sibling, pre- ferably the next older or next younger sibling, were in- terviewed. Twelve families in each of the three disease groups were studied. The psychological variables studied were (i) anxiety, (2) depression, (3) guilt, (4) future time perspective, (5) adjustment, and (6) Openness of the family in dis- cussion of the illness. Psychological functioning of the patients and siblings was assessed using a battery of psy- chological tests. This included the General Anxiety Scale for Children, the Incomplete Sentences Test (which measures future time perspective), The California Test of Personality (which measures adjustment), the Thematic Apperception Test Andrea Farkas and the Waechter Projective Pictures (for ratings of guilt and depression), and the Family Relations Test (which mea- sures intra-familial affect). The mother was interviewed to obtain a devel0pmental history of the sick child, demo- graphic information about the family, and a general impres- sion of how the family had adapted to the child's illness. Ratings of the Family's Openness about Discussion of the Illness were made by an independent rater on a five-point rating scale from tape recordings of the interview with the mother. It was found that the families of children with ter- minal illness adapted as well as the other two groups. It had been expected that children with terminal illness would show more anxiety, guilt, and depression, and have a shorter future time perspective and lower adjustment scores than children with chronic or less severe illnesses. Siblings' psychological functioning had been expected to vary simi- larly, depending on the severity of their brother's or sister's illness. Further, it was expected that in fami- lies where the illness was more Openly discussed, the pa- tients and siblings would experience less anxiety, depres- sion, and guilt than in families where such discussion was less open. These expectations were not substantiated, with the exception of the finding that children from families which Andrea Farkas discussed the illness more openly did express less guilt than children from less open families. There were no sigé nificant differences in anxiety, depression, guilt, length of future time perspective, or adjustment between the three groups of patients with illness of varying severity, or between sibling groups. No relation was found between the children's level of anxiety or depression and the family's openness in discussing the illness. The limitations of applying the conclusions from this research to other families of_sick children are discussed. Possible explanations are offered for the failure of the study to find psychological differences among groups of children with illness of varying severity. Finally, sug- gestions are made for further research into psychological adaptation to physical illness by children and their families. Dissertation Committee Dr. Donald Grumman, Chairman dsz4figkf§7 Dr. Lucy Rau Ferguson Date I Dr. Lawrence Messe Dr. Albert I. Rabin ADAPTATION OF PATIENTS, SIBLINGS, AND MOTHERS TO CYSTIC FIBROSIS BY Andrea Farkas A DISSERTATION Submitted to Michigan State University in partial fulfillment of the requirements for the degree of DOCTOR OF PHILOSOPHY Department of Psychology I973 DEDICATION To Dr. Harry Shwachman, his patients, and their families and to my parents, Joseph and Sephie Farkas ACKNOWLEDGEMENTS I am deeply grateful to Donald L. Grummon for teaching me about clinical psychology and research, for his encouragement of this project, and for his unconditional positive regard throughout these years. I would like to thank Dr. Lawrence Messe for his much appreciated advice on statistical matters, Dra. Lucy R. Ferguson and Albert I. Rabin for their critical comments and constructive suggestions, and Drs. Richard Schnell and Richard Pomerance for many helpful discussions. I am particularly grateful to Dr. Harry Shwachman for making this work possible, and to Drs. Kon-Taik Khaw and John Lloyd-Still, Miss Bonnie Oliphant, Mrs. Virginia Rice, and Mrs. Julie Henry for assisting me in the selection of subjects for the two cystic fibrosis groups. I also wish to thank Dr. Kenneth Gabbay and Mrs. E. Salaba of the Adolescent Diabetic Clinic for providing access to diabetic patients. My thanks to Mrs. Elizabeth Geist and Miss Ellen Lehn for their cheerful assistance as raters, and to Drs. Hallowell Churchill, Lois Eichler, and Cathy Widom for their generous loan of calculating equipment. iii Finally, I wish especially to thank Jochen for his continuing support and warm encouragement, particularly during the final stages of this research. This research was supported by NIH Grant MH 49909-Ol. iv TABLE OF CONTENTS Chapter Page I. INTRODUCTIONeeweeaeeeeeeaeeeeeeeeaeeeeeoe000000000' ResearCh 0n thQ Dying Chi'daaaeeeeaaeeeeeeeecee 3 Effects of a Child's Fatal Illness on Family MEMDEVCeeeeeeoeeeeeeeeeeeeeeeaeeeeeeeeeeaeaee 8 Previous Psycho|0gical Research on Children WIth cyStIC FibrOSIBaeeeeeeeeeeeeeeeeeeaeeeee'5 This R989.rCheeeeeeeaeeeeeeeeeeeeaeeeeeeeeeeeee'7 HypOthCSQSeeeeeeeeeeeeeaeeeeeeeaeeeaeeaeeeeeeea2 ‘1. METHOD........................C...................26 Limitations in Selection of Subjects...........26 SUbJQCtseeaaeeeeeeeeeeeeeeeeaeaeeeeeeeeeeeecaee3o Procedureseaeeeaoaeeeeeaaeeeeaee0000000000000003 StatISt'cal Ana'YSISaeeeeeeeeeeeeeeeaeeaeeeeeeesh III. RESULTSC..........O......l.........................55 seet.0n AS Original HypOthESCSeeeeeeeeeeaeeeeess ANX'EtYeeeeeeeeeeeeeeeeeeeeaeeaoaeeeeeeeeeaaSS DeprQOS‘oneeeeeeeeaeeeeeeeeeeeeeeeeeeeeeeeee58 Adjustment..o..............o................59 GUT'taaeeeeaseaeeeaeeaeaeaeeeeeeeeaeaeaeeeee6' FUture Time Per8peCt‘Veeeeaeeeeeaeeeeeeaaeae6l Openness in Discussion of the Illness in the FaMI'Yeeaeeeeeeeeaeeeeeeeeaeeeaeeaeeae62 Section 8: Analysis of Related Data...........65 ANX‘Qtyaeeeeeeaeaeeeeeeaeeaeeeeeeeeeaaeeeeee L'Q SC‘|eaaeeeeeeeeeeeeeeaeeeeee000000000066 Sex DIforenCQSeaceeeeeeaeeeeeeeeeeeeeeeae68 Comparison with GASC Scores from WSQChter's StUdYeaeeeaeoeeeeeeeeeeeeeeeeGg corre|8tion3 “'th Other VarIab'CSeeaeeeeee7' Depression..................................72 Future Time Perspective.....................75 overprOtQCt‘ONaeeeeeeeeeeeeeeeeaeeeeeeeaeeee78 Negative AffeCteeeeeeaceeoeeeeeeeeeeeeeeeeeaB' Early Developmental Disturbances............84 P8YChO|Og'C'. symptOMSeeeeeeeeeeeeaeeeeeeeeea6 Maternal Variables....o.....................88 IV. DIscusSIONO0I.OOOOOOOOOOOOOOOOOOOOOOO00.0.0000000093 Suggestions for Further Research and Greater Involvement of Psychologists in Medical ClINICQeeeaeeeeeeeaeeeeeaeeaeeeeeeeseeaeaeee'02 TABLE OF CONTENTS (continued) Chapter Page v0 SUMMARYOO0.00000000000000000.000000000000000...'06 BIBonenAPHYOOCOO.00000010000000.0000.00000000000000000.0I08 APPENDIX A: Letter to Parents Explaining Purpose of Study and Tasks Invoived.................ll5 8: Number of Children per Family, Number of Children with Illness, Religion, and In- come in Thousands for Families...........ll6 C: The General Anxiety Scale for Children.....ll7 03 TP INCOMDIEte SENtenCESaaeeeaaeeaaaeaeeeeee|'9 E3 WHEChter PrOJBCtiVE Pictures...............'20 F: DEDFGBSIOD Scale...........................|24 G3 GUI't SCR'EeeeeeeeeeeaeaaaeeaaeaeaeeeeeeeeeI25 : Overprotection Items From the Family Re- 'at'0n3 TQSteeoeeeaoeeeeeeaeeeeeaeeeaeeaaI27 1: Negative Affect Items From the Family Re'atIons TQSteeeeaaeeaeaeeeeeaeeeaeeaeea'28 J3 Maternai INterVi9' SChedU'eeeaaeeeeeaaeeeaa'29 K: Openness of the Family in Discussion About The 1"n938 Rating sca|eaaaeaeeeeeeeeeaaa'42 L: Warmth of Mother-Child Relationship Rating scalesaeaeeeeeeeeaeeeeeaeeeeeeeeee'43 M: Mother's Depression Rating Scale...........l44 N: Mother's Judgment of Sick Child's Mood Rating Scale.............................IAS O: Level of Burden on Mother Rating Scale.....l46 3 DISC‘DIIHQ Rating sca'eeeeeeeeeeeeeeeeaaaee'47 vi Chapter APPENDIX Q: R: 8: TABLE OF CONTENTS (continued) Page Early Developmental Disturbances...........l48 PGYChOlog'CB' Symptoms....o................I49 Raw Data: Patients' and Siblings' GASC Scores, GASC Lie Scale Scores, Depression and Guilt Ratings, Incomplete Sentences Test Scores, Total Adjustment Scores from the California Test of Personality, Ne- gative Affect and SELF-SIB Overprotection Scores from the Family Relations Test, Number of Early Devel0pmental Distur- bances, and Number of Psychological SymptONSaeeeeeaeeeeeeaeeeeaeeoeaeeeeeeeeea'50 Raw Data: Ratings from the Maternal Inter- view of Openness of the Family to Dis- cussion about Illness, Maternal Warmth to the Sick Child, Maternal Depression, Level of Burden on Mother Caused by the Illness, Mother's Judgment of the Sick Child's Mood and Mother's Judgment of the Sick Child's Need for Discipline Relative to Sib"nQSeaeeaaeeeeaeeaeeeeeaeeeeaeeeeee'56 vii Number 2. 3. 5. 7. LIST OF TABLES Page Age, sex, age at diagnosis, number of hospi- talizations for patients and age, sex and rCIEtIVQ age Of 8Ib'In93aaeeeeeeaeeeeeeeaaeaeaeeeB' PrOJECt'Ve Test P'Cturesaaceeeeaeeeeeeeeeeeeeeeeaeehs Table of means and standard deviations for patients' scores on the GASC, Depression and Guilt ratings, Total Adjustment scores of the California Test of Personality, the Leasing Incomplete Sentences test, GASC Lie Scale, SELF-SIB Overprotection score, Negative Affect to Sibling, Early Develop- mental Disturbances, and Number of Psycho- |Og‘ca' Symtpoms.............o...................55 Table of means and standard deviations for siblings' scores on the GASC, Depression and Guilt ratings, Total Adjustment scores of the California Test of Personality, the Leasing Incomplete Sentences test, GASC Lie Scale, SELF-SIB Overprotection score, Negative Affect to Sibling, Early Develop- mental Disturbances, and Number of Psycho- 'Og‘ca. symptOMSQQeaeeaeeecaaeeeeeeeaaaeeeaeaeeeas7 Mean ratings of Maternal Openness to Discuss Illness, Warmth, Level of Burden, Judgment of Sick Child's Mood, and Sick Child's Need for Discipline taken from Maternal Inter- V'eHOOOOOOOOOOOOOOOOOOOOOOOOOOOOOOOO0.00.00.00.0059 F scores from 3 x 2 x 2 ANOVA with repeated measures on patients and siblings for anxiety, depression, total adjustment scores on Calif- ornia Test of Personality, guilt, and future time perspective scores on Incomplete Sentences TestOOOOO0......00......000......00.0.0000000000060 F values from Simple ANOVAs on Maternal V3f'8b'980eeeeeaeaeeeeeeeeeaeeeeeeeeeeeeeeeeeaaooG} viii Number 8. 9. IO. l2. I3. l4. l5. l6. LIST OF TABLES (continued) Page Correlations between ratings of Family Open- ness in Discussion of Illness and Patient and Sibling scores for Anxiety, Depression, AdJUStmENt, and GUT'teaaaaaeeeeeeeaeeeeea0000000063 F Scores from a 3 x 2 x 2 ANOVA with Nested FaCtors for GASC Lie Scores......................66 Mean GASC Lie Scale Scores, Standard Deviations, and Ranges for Patient and Sibling Groups........67 Mean GASC Anxiety Scores (and Standard Devia- tions and Ranges, where available) for Male and Female Patient and Sibling Groups in this Research and for Normal Males and Female8..........................................68 Mean GASC Scores and Standard Deviations for Patients in the CF', CF2, and DB groups and in Waechter's I968 Study of Children with Fata', Chronic, and Brief 1'Inessaeeeeeeeeeoeeeee68 Correlations Between GASC Scores and Patients' Number of Psychological Symptoms and Patients' and Siblings' SELF Overprotection Scores.......o...................................7| Mean Depression Scores, Standard Deviations, and Ranges for Patients and SIb'inQSeeeaeeeeeeeae73 Depression Ratings, Standard Deviations, and Ranges for Mothers Taken from Recordings of the Maternal Interview, and Correlations with Patient and Sibling Depression Ratings Taken from the prOJeCt‘VQ TEStse00000000000000.0073 Future Time Perspective Expressed in Years Incomplete Sentences Test Scores, Standard Deviations, and Ranges of Scores for Patients and Siblings in this study and Mean Incom- plete Sentences Test Scores for Female Sub- JECtB in L983‘ng'8 I972 StUdyeeeeaeeeeeeeeeeeeeaa76 ix Number l7. l8. l9. 20. 2|. 22. 23. LIST OF TABLES (continued) Page F scores for 3 x'2 x 2 ANOVA with nested Factors for SELF-SIB Overprotection scores from the Family Relations TESteaeeeeeeeeeeeeeeaeo79 Mean SELF-SIB Overprotection Scores for Older and Younger Patients and Siblings................80 Mean Negative Affect Scores and Ranges form the Family Relations Test for Patients and Sib'in980aaeeaeeeeeeeeeeaeeeeeeeeeeeeaeaeeaeea00.82 Early Developmental Disturbances Reported by MOthers Of Patients..............................85 Correlations between Number of Early DeveIOp- mental Disturbances and-Number of Psycholo- gical Symptoms for patients, Maternal De- pression and Maternal Rating of Level of Burden Caused by I.I"€33eaaaaeaeeeeaaaeaaeeeeeee.85 Psychological Symptoms of Patients in the Three Disease Groups as Reported by their Mothers and Differences between means as obtained from simple ANOVAeaaaaaeeeeaeaeeeeeeaeeaeeeeeeeeaa7 Mothers' Reported Sources of Emotional Support.....9l CHAPTER ONE INTRODUCTION In recent years there has been a growing awareness of the psychological needs of dying patients, their families, and the hospital staff who care for them. All are involved in a network of complex, emotional inter-relationships as they strive to prevent, for as long as possible the pa- tient's death. When that patient is a child, the effect on the family and staff is, in most cases, even more intense. For the death of a child marks, even more than the death of an adult, our failure as parents, doctors, nurses, and other helping professionals to nurture and sustain life. The effects on a child of having a terminal illness and the effects on family members of living with a fatally ill child have received little attention. The present research concentrates on the effects on child patients, siblings, and mothers. [it is intended to fill a gap in the research done to date on family adaptation to fatal illness:] The emphasis in past studies has been on parents' reactions with occasional and fleeting mention of the effect on siblings. The author. has seen in her clinical work the devastating effect that the death of a sibling can have on the deveIOpment of the surviving children. Other researchers have suggested that the experience of death of a sibling in childhood can be a contributing fac- tor to the devel0pment of schi20phrenia (Rosenzweig and Bray, I943; Moriarty, I967) or can lead to severe phobias, hysteri- cal somatization, delinquency and school problems (Binger et al., l969; Cain, Fast, and Erickson, I964; Cobb, I956). For many children the effects are not as serious, but there are, it is hypothesized, some effects on all children. The present research is aimed at trying to define what these general effects are and at trying to determine what factors make the terminal period more or less disruptive for a child. Hopefully if we are able to learn something about how children react to this experience we can begin to develop ways of helping them to cope more effectively with it. This research deals with families in which one or more children have cystic fibrosis. Cystic fibrosis is an inherited disease producing chronic involvement of the respiratory and digestive systems. It is transmitted as a recessive trait from both parents and occurs approximately once in every I600- 2000 live births. It can be diagnosed from birth, although in many children the diagnosis is not made until years later. At present cystic fibrosis is incurable, although available treatments can prolong life into the teens and sometimes into the twenties. This disease, then, necessitates long-term family adaptation, different in many respects from the type of adaptation that occurs in families of leukemia patients, who have been most frequently studied. Review of the Literature figgearch on the Dying Child The literature on the dying child consists mainly of. brief case reports and advice from physicians and nurses to their colleagues on management of the dying child and his parents (Murstein, l958; Solnit and Green, I959; Tisza, I962; Williams, i963; Verwoerdt, l966; Green, l967; Rothenberg, I967; Easson, I968; KoOp, i969; Lascari, I969; Crammond, i970; Goldfogel, l970; Schowalter, i970; Nervyn, l97l). In an ef- fort to make up for the lack (until very recently) of medical and nursing courses on the care of dying patients, doctors and nurses with some awareness of the problems have generalized from their experiences to teach others. These articles pro- - vide guidelines for dealing with the issues of how, when and to whom to deliver the diagnosis, how to provide personalized care, especially in teaching hospitals, and how to aid the parents in management of the child's daily life. These articles stress the importance of clear, continuing communi- cation between the doctor and the parents and the maintenance of a realistic, yet hopeful, position. As to what the child should be told, the most consistent advice is that the medical staff should be guided by the questions the child asks and by his age, although some doctors feel strongly that the seriousness of the illness should be hidden from a child even if he asks about it (Winer, l970). The medical staff are warned to expect expression of irrational anger or guilt from the parents. And the doctors and nurses are encouraged to express and share their own feelings about death and to uti- lize support from other hospital staff if it is available. These articles deal in generalities and are probably most helpful in giving doctors and nurses permission to confront patients and their parents more honestly and directly, rather than in teaching them specifically how to deal with indivi- dual cases. ITJttie psychological research has been done on dying children and their families. Partly this may be because death has been treated as a taboo subject\during the Twentieth Century much as was sexuality ithhe Victorian era (Gorer, i965). This taboo about death is slowly lifting and the number of thanatological research studies is increasing, although resistance to such studies by the medical staff who control patient populations is still fairly common. As restrictions on interviewing patients are lifted, the second factor which inhibits research in this area be- comes increasingly important. That is the emotionally exhausting nature of a project which requires many hours of direct contact with children who are near death. Just as the medical staff, the family, and the dying child must come to grips with feelings of despair, depression, and hopeless- ness, the psychological researcher similarly must face his or her own reactions to the experience of knowing a dying child and should seek support from available sources. Some 5 thanatological research units have established weekly meetings solely for the purpose of discussing the feelings of the re- searchers (Williams and Vollman, l97l). Herman Feifel, one of the earliest thanatologists, says of work with the dying, ”Few undertakings in psychological research are more emo- tionally exacting. Pain and death are themes not comfortably encompassed by categories of methodological rigor and theoretical relevance.'(Feifel, l966). Feifel's remarks suggest a third reason for the scarcity of work in this area. The complex emotional reactions, both conscious and unconscious, of peOple to their own impending death or to the death of a loved one are not easily measured by existing tools. One task of researchers currently working in this field is to define apprOpriate regions of investiga- tion and then to improve the methodology. Despite the difficulties, some research has been done on dying children and their families. The central issue of this research has been the nature and extent of the death anxiety these children experience. The majority of authors studied leukemic children, although Waechter's (i968) group included children with a variety of fatal illnesses and McCuliy (l963) used a population of children with muscular dystrophy. Morrissey (I964) and Natterson and Knudsen (i960) both observed and interviewed hospitalized leukemic children and gained further information from their medical charts or reports of social workers' interviews with the children's parents. Both studies concluded that, with few exceptions, anxiety about death is predominant over other worries only in children over l0. Some disagreement about this issue is voiced by Vernick (l965) who feels that when an atmosphere of honesty and freedom to question is created on a pediatric leukemia ward that the near-universality of children's fears of death can be observed. Waechter (l968) studied the anxieties of fatally ill children in some detail using both a measure of generalized anxiety, the Sarason General Anxiety Scale for Children, and ratings of more specific forms of anxiety as expressed in a modified TAT administration. Her comparison groups consisted of children hospitalized for chronic and brief, but not fatal, illness. The fatally ill children had general anxiety scores double those of the children hospitalized with brief or chronic illness. She found that while loneliness imagery was expressed more often by children under eight, there was little difference in the expression of fantasy concerning death or mutilation among fatally ill children in different age groups. YMCCuily (I963) also studied fantasy in fatally ill children. Ne found that children with muscular dystrophy (age lO-IA) differed from healthy, normal children in that their projective responses showed more imagination and less reliance on the stimuli, more concern with death and disaster, and more qualified and .- unrealistic outcomeSA‘ A comparison between the fatally ill in. or group and a second control group of children with polio showed that the polio controls tended to have a greater ac- ceptance of catastrophes as natural events and had more realistic resolutions in stories involving the future. Murstein (l958) compared two sessions of psychological testing of a twelve-year-old leukemic boy, one done soon after diagnosis and the other in the terminal phase. While no significant changes in intellectual functioning were noted, the projectives reflected a progressive weakening of hope and ambition as well as primitization of response. While this case was atypical in that the child's mother visited him only once in his four-month hospitalization, it does point up the need to consider the psychological effects of long-term hospitalization and serious illness on children. There are several articles which fall between the categories of research reports and case studies. They are reports of intensive observations of a fairly large number of hospitalized fatally ill children. Richmond and Waisman (i955) report that the fatally ill children they observed rarely showed overt concern about death, but seemed to have an air of passive acceptance and melancholia.‘§Schowalter (I970) describes four developmental stages in the child's reaction to terminal illness. As the child becomes older, his conception of death becomes more appropriate and stable, his awareness of his own fate increases, and guilt, anger, and fear become more significant components of his emotional reaction.{ .\ ,l _ffects of g;Child's Fatal Illness on Family Members The anticipation of the death of a child from a fatal illness occurs within the context of a family, as well as within the context of a hospital, and has repercussions on all family members. In contemporary American society, where children are expected to outlive their parents, the antici- pated death of a child disrupts the normal pattern of daily existence and undermines the basic philosophy of hope for an ever-brighter future. Research studies on the family members' reactions to the presence of a terminally ill child have in- creased in recent years, but have concentrated on parental reactions and only slight mention has been made of the effects on siblings. Most of the research has been based on parent interviews, although in the Boseman et al. (I955) and Murstein (I960) studies, some projective and other testing was done. Most of the studies report extensively on the parents' feelings of guilt, inadequacy, and self-blame for their child's illness (Richmond and Waisman, l955); Friedman, Chodoff, Mason, and Hamburg, l963; Gardner, l969; Evans, Saunders and McCarthy, l968). Reactions to the diagnosis ranged from general accep- tance (Friedman et al., l963) to disbelief and denial in the parents as seen by Bozeman, Orbach, and Sutherland (I955) and Natterson and Knudson (l960). Remission was an occasion for renewed hope for almost all parents. When remission ended and hospitalization became necessary, separation was noted as a special problem. Relatives and friends were not generally supportive or help- ful, partly because they expected conflicting behavior from the parents, i.e. parents were expected to be grief stricken and yet not to have given up hope for their children (Friedman et al., l955). Other parents of leukemic children were seen as the greatest source of comfort (Bozeman et al., l955; Friedman et al., I963). Mothers experienced a symbolic threat to their own existence through their close identification with their children (Natterson and Knudsen, I960). Both parents frequently experienced a reawakening of feelings related to earlier losses (Orbach, I959). KIT parents were allowed to participate in the care of their children they generally felt Igggmguilty(Richmond and Waisman, I955: Hamovitch, l9655§> Parents were caught in a conflict between protecting the child and motivating the child for possible growth and tended to overprotect their child (Tisza, I962). Anticipatory mourning in these parents involved two con- flicting tasks, i.e. (l) the gradual relinquishing of the . child and, (2) caring for the child in all of the special ways necessitated by his illness (Futterman, Hoffman, and Sabshin, I970). Futterman et al., (l970) see anticipatory mourning as including five stages through which most parents pass. These are: (I) acknowledgment, in which the parent struggles with hepe versus despair: (2) the experience of grief which shows peaks at diagnosis, the first relapse, and in the terminal phase but which modifies and softens the eventual death 10 (3) resignation and reconciliation; (4) detachment which includes the redirection of energy to other family members; and (5) image fixation in which the conscious mental repre- sentation of the child is molded into a relatively permanent form. Futterman et al. (i970) says of anticipatory mourning: Our experience suggests that this may help parents deal effectively and supportively with the siblings of the lost child. Hope- fully, if the aspects of anticipatory mour- ning can be identified and parents can be supported in their mourning work, turmoil, family disruption, pathological reactions and disturbed family relationships might be prevented. Several articles point out the psychOIOgicai difficulties that may result from losing a sibling in childhood; all are retrospective studies. Binger et al. (I969). interviewed parents in 20 families who had lost a child from leukemia. In half of the families one or more previously well siblings showed behavior patterns indicative of difficulty in coping with normal tasks of childhood. symptoms included enuresis, headaches, poor school performance, school phobia, and severe separation anxieties. Guilt reactions were common, as were fears that the sibs would contract the same disease that had claimed their brothers or sisters. From the interviews it was ascertained that both parents and siblings had shown an- ticipatory grief reactions during the terminal phase of the child's illness. Perhaps the most interesting and startling finding was that in one half of the families, one or more family members who had not previously been in treatment required 11 psychiatric care, including several who were hospitalized for depression. Dr. Albert Cain and his associates at the University of Michigan have reversed the pattern of investigation and have looked through psychiatric clinic records to find children whose symptoms reportedly%stemmed, at least in part, fme~losseof_a sibling (Cain, Fast, and Erickson, I964; Cain and Cain, l964). These children showed symptoms of de- pressive withdrawal, provocative behavior, many forms of acting out, and poor school performance. The children were fearful of losing control and saw themselves as monsters or potential killers.) They had distorted concepts of illness, death, doctors, hosgitals, and religion. Many suffered from death phobias and were fearful of dying at the same age as their siblinng Disturbances in cognitive functioning, es- pecially around ideas of time and causality were noted. A special burden was placed on a child whom the parents consciously or unconsciously deemed as a replacement for the dead sib. These children were often heavily overprotected and themselves became quite phobic and frequently were pre- occupied with death and funerals. Hysterical somatization was observed as these children identified with their dead sibling in a hopeless struggle to compete with their parents? idealized image of the dead brother or sister. It should be noted that these reactions occurred among a psychiatric clinic population and would not necessarily be the reactions of all 12 normal children to the death of a sibling. Cobb (l956) mentions the school problems encountered by the remaining sibs and notes that often they had learned little in school during the time their brother or sister was ill. Hilgard (I969) reports on case histories of a suicide, a case of psychosomatic symptom formation, and a paranoid epi- sode as reactions In adults to the anniversaries of sibling deaths in childhood. These reactions were noted to be much more severe than the reactions these people had at the time of the traumatic death. There are a few scattered reports in the literature on psychotherapy done with children around the issues of losing or having lost a sibling. Feinberg (l970) reports on ten months of preventive work done with two sisters, aged 7% and 9% whose six-year-old brother was diagnosed as having leukemia. Therapy was initiated to help the girls adapt to their brother's expected death, despite the fact that they were not showing any psychiatric symptoms. Feinberg comments on the importance of the therapist's truthfulness with the children. The necessity for "immunizing" discussions about other deaths the children have experienced and for expression of the natural guilt and anger the children feel is stressed. Initiation of mourning, i.e. the sharing of memories about the deceased, was another goal of treatment. Feinberg agrees with Wolfenstein (I966) that pre-adolescent mourning should be differentiated from adult mourning. He concludes with a wish for increased 13 “receptivity on the part of the (psychiatric) profession for preventive intervention with all bereaved children, sympto- matic and asymptomatic.” Another report of brief therapy describes treatment of a 6% year old boy who six months previously had lost a sister, age 2%, of asthma (Rosenblatt, I969). The boy's symptoms in- cluded hysterical wheezing and preoccupation with death. Lifting of the repression by clarifying the boy's under- standing of religious retribution allowed him to manifest his mourning. In contrast to Feinberg, Rosenblatt expressed some skepticism that emotional illness could be prevented in children as a response to loss. Zeligs (l967) freed a five- year-old boy from depression, sleep phobia, and daydreaming in school with two interviews of open discussion about the death of his one-year-old brother. Norman Paul (I965, I967) believes that the encouragement of grief reactions is a prOper goal of family therapy for be- reaved families and one which has been severely neglected. Unresolved feelings about loss can lead to maintenance of per- vasive defenses against further loss and disappointment which can lead to fixated family equilibriums like those frequently found in neurotic and schizophrenic families. The literature on children's mourning is rather contro- versial. The controversy centers around the issue of whether or not the quality and duration of children's mourning is similar to that of the adult. Melanie Klein believes it is l4 (Klein, I940). Bowlby (I960, l96l, l96la) prOposes a three-stage process of protest, despair, and reorgani- zation or detachment. He emphasizes the similarity between experiences of separation and grief and espouses the theory that by age six months the child has deveIOped the capacity to mourn. The course and duration of the mourning varies, however, with the age of the person involved. Bowlby be- lieves that children retain the image of their mother for several weeks at least. He also emphasizes the importance of these early experiences on future development (l96la, I964), believing that mourning in early years is, ”unfavorable to future personality deveIOpment and thereby predisposes to psychiatric illness (Bowlby, l96l).” Anna Freud disputes the ability of a six-month-old child to mourn and believes that in a child between one and two years mourning takes place for only 36-48 hours (Siggins, I966; Bowlby, l960). Furman (I964) feels that a child of 2 or 3 is capable of understanding death as an external reality while a 3% to 4 year old is capable of the internalization of ob- jects and is therefore able to mourn as an adult does. Wolfenstein (I969) believes the main difference between child and adult mourning is that children transfer their feelings more easily to new objects and that therefore for them there is ”no protracted absorption in the work of decathecting the lost object.” Rochlin (I959, I967) believes that children experience neither depression nor elation and are thereby 15 unable to mourn. Identification with the lost object forms the keystone of a system of defense mechanisms which Rochlin terms the ”loss complex.” Instead of showing the usual signs of adult mourning, the child withdraws and regresses to the point where identification with the object was fixed. Helene Deutsch believes the ego of the child is insufficiently de- veloped to be able to bear the mourning work. Narcissistic self-protection is afforded by either infantile regression expressed as anxiety or omission of affect (Deutsch, I937). Though referred to in articles on families of dying children, no study of anticipatory mourning in children comparable to Lindemann's (I944) study of mourning in adults has yet been conducted. Previous Psychological Research on Children with Cystic Fibrosis The psychological aspects of the care of children with cystic fibrosis have been dealt with in a number of articles. The implications for parents and families are expressed by one doctor who states, "In the whole of paediatrics there is no chronic illness which merits long-term parents' support more than cystic fibrosis" (Pinkerton, I969). Problems for families include managing the financial strain of having more than one cystic fibrosis child, handling the sibling jealousies about the amount of parental time spent in care of the sick child, and disciplining the sick child in spite of his illness (Belmonte, I967; Burgess, I969; McCoIlum and Gibson, I970). Some parents attribute children's school adjustment problems, 16 learning problems, and delinquency to the strain of the disease (Tropauer, et al., I970). Meuwissen (I97l) stresses the importance of paying more attention to what she calls the “forgotten siblings' to help them deal with their jealousy and anger and thus to prevent further family disorganization. Talking to the children about their illness is another problem that must be faced. Waechter (I968, I97l), in studying the death anxiety of fatally ill children, found that at least some of the parents of cystic fibrosis children were more able than parents of children with leukemia or other carcinomas to discuss treatment aspects of the disease realistically. Some cystic fibrosis families, however, maintain a "web of silence" around the illness which increases the burden of frustration felt by all concerned (Turk, I964). As with other fatal illnesses, the incidence of guilt, denial, and recurrent depressions in parents of cystic fibrosis children Is high, but Tropauer et al. (I970) notes, too, that we have no norms by which to judge the behavior of parents of chronically ill children. Tropauer et al. found in the 23 families they studied that, ”Where openness and mutual support prevailed, the child's illness had little disruptive impact on the family unit.” Where children were uncooperative about their treatment, they often found marital problems and lack of familial closeness. ’ Because children with cystic fibrosis are now living longer (Shwachman, Kulczycki, and Khaw, I965; Shwachman, l7 Redmond, and Khaw, I970), there are new problems to be dealt with in families of adolescent cystic fibrosis patients. The adolescents see themselves as being different from and in- ferior to their peers. They resent the dependency necessi- tated by the cystic fibrosis treatments, and they protest against the unfairness of having the disease. They may be- come depressed or hypochondriacal and may sabotage their treat- ment (Pinkerton, I969). In addition to the normal difficulties of adolescence, these children have worries about being unable to get a job because of their disease (Patterson, I969) or not being able to marry and have normal children, as well as, of course,the Iimitied life span they face. This Research Clearly there is still much to be learned about how families adapt to having a child with cystic fibrosis. This research aims to identify how siblings are typically affected by the illness, to increase our understanding of the experience of the terminally ill child, and to identify which kinds of families tend to have a particularly difficult adjustment. This project examined families of children who are at two different stages of the disease. Cystic fibrosis affects children with varying degrees of severity in earlier stages. Some children can lead nearly normal lives except for taking medicines and attending yearly clinics. Other children, usually those closer to the terminal phase of the disease, 18 are bed-ridden, cannot attend school or lead active lives, have frequent visits to the doctor, plus a heavy daily rou- tine of medicines, physical therapy, and aerosol treatments. The group Is composed of families of severely ill children who are in or approaching their terminal phase. The ill child himself, his mother, and one or more of his siblings between the ages of 7 and 2i were included in the research. Pre- ferably the next older and next younger siblings were studied. If either of these siblings himself had cystic fibrosis, another normal sibling was studied, If available. All sub- jects are patients or siblings of patients at the Children's Hospital Medical Center, Boston, Massachusetts. The designa- tion of extent of illness has been made by the doctors and nurses of the Children's Hospital Chronic Nutrition (Cystic Fibrosis) Clinic. Control groups include both families of cystic fibrosis patients at earlier stages of the illness and families of a group of chronically, but not fatally, ill children. Diabetic children were chosen for the latter control group since, like the CF children, they require long-term care, much family in- volvement, and continuing contact with doctors and hospitals. The CF control group includes families of children who have a mild course of the disease throughout and have had no serious hospitalization. In all control groups, the affected child, mother, and one or more normal siblings in each family were studied. The expectation was that the more intense the I9 family's experience with the illness,the more they will re- semble families of the terminally ill children. Families of diabetic children will be expected to be affected by their child's illness and hospitalizations but to a lesser extent than the families in the main experimental group who must, in addition, come to terms with the eventual death of their child. Because of the relatively small number of subject families of fatally ill children available for this research, this study concentrated on variables which seemed likely to be of major importance. Since the general assumption that was examined was that patients and siblings of patients are affected by their experiences with illness in ways that dis- turb normal development, we have chosen to look at several levels and measures of the children's general psychological health. While studying the children's overall level of per- sonal and social adjustment, we also looked at more particular measures such as anxiety, guilt, and depression. In addition this study hopefully has added to the literature on children's ideas of future time perspective by revealing how the ex- perience of living with a fatally ill child affects the deve— lopment of this concept. A fuller justification for the study of these particular variables is as follows: Anxiety ”One thing common to all patients is anxiety connected with the diagnosis of their condition” (Duffy and Hansen, I969). 2O Certainly from the time of the diagnosis onward, a fatally Ill child and his family have much to worry about. Fears of mutilation and threats to body integrity, fears of se- paration, loneliness, and social isolation, and, eventually, fear of death are all likely causes of anxiety. Waechter (I968) found that hospitalized fatally ill children had anxiety scores (on the Sarason General Anxiety Test for Children) double those of children hospitalized with brief . and chronic, but not life-threatening, illness. The fatally ill children's scores were three times the norms reported by Sarason (Sarason et al., I960) for normal children of the same age. 0n projective tests she found that children with cystic fibrosis tended to have somewhat lower anxiety scores than other fatally ill children. There are also obvious anxieties connected with being a member of the family of a fatally ill child. The special care of cystic fibrosis patients arouses continuous anxieties. The parents' anxieties about their child's prognosis could hardly be kept from being communicated to other family mem- bers. The separation from a hospitalized sick sibling as well as separation from parents while they visit the sick child is a further cause of anxiety in the family. Reality- based fears of being a carrier of cystic fibrosis with the possibility of having children with cystic fibrosis gives siblings extra cause to worry. Anxiety, then, would appear to be an Issue of central importance to these families. 21 Adjustment The term adjustment denotes a person's ability to suc- cessfully adapt his behavior to the stresses he encounters. Having cystic fibrosis presents the patient (and other fa- mily members) with a continuing series of stresses. Espe- cially if it goes undiagnosed through a child's early years, cystic fibrosis can cause extreme difficulty with feeding and toilet training, thereby disrupting and burdening the mother- child relationship. In general It seems likely that children with cystic fibrosis would find adjusting to the normal tasks of childhood and adolescence more troublesome than healthy children. This expectation has been borne out by the cases reported (Tropauer, I970; Patterson, I969; Pinkerton, I969). Similarly, siblings of children with cystic fibrosis may also be handicapped by having to share much of their parents' affection and attention with a brother or sister whose medi- cal condition necessitates constant care. Mothers of fatally ill children often express the feeling that they are short- changing their healthy children because of their devotion to the child who is sick. Bowlby (l964) and others (Paul, I965, I967; Cain, Fast, and Erickson, I964) have pointed out that early experience with loss frequently diminishes one's capacity to adapt to later losses or disappointments and increases the rigidity of a person's defensive organization. It should be pointed out that the “loss” of a chronically ill brother or sister as an 22 active playmate and companion often predates the child's actual death by some time. This factor, plus the anxiety engendered in all family members by the illness and antici- pation of the child's death, lead to the expectation that siblings of terminally ill children will show many of the distortions in personality organization which have been noted in children following the death of one of their sib- lings. Thus we would expect the ceping mechanisms of the siblings in our experimental sample to be less well-developed than those of siblings of controls. Guilt and Depression It is hypothesized that the presence of dying children in a family intensifies the sibling rivalry feelings of the healthy siblings. Such rivalry is hypothesized to be the re- sult of extra attention which the ill child receives. This attention is a consequence of several factors. ‘One factor is .—..-._. - the reality demand for special treatment,(frequent trips to W4 the hospital, etc. Also, the parents' guilt leads them to P‘ \ ngvlsh more affection than usual on a child who has a limited life span::TThe healthy children experience intensified anger directed {Swards the ill child as well as intensified guilt. The guilt may in part be a result of that anger and in part the result of feeling responsible for having caused the ill- ness. This fantasied sense of responsibility has been noted in children who have lost a sibling by Cain, Fast, and Erickson (I964) as well as by the author in her clinical work. Also, 23 guilt is usually seen as one part of the anticipatory mour- ning response and thus would be expected to be found es- pecially in families of terminally ill cystic fibrosis children. Depression is similarly an important part of anticipa- tory reactions and one which has been reportedly found in parents of chronically ill patients (Binger et al., I970). Depression would be expected to also be found in the sib- lings, expecially siblings of the terminal patients. Time Perspective The structuring of time takes on special importance for the members of a family with a fatally ill child. The under- lying question of ”How long will this child live?“ is a major influence in determining the context in which the family mem- bers deal with one another and with the medical staff (Glaser and Straus, I965). To the healthy children in these families who are in the process of formulating their ideas of time - past, present, and future - and of death, the presence of a fatally ill child may create some unusual complications. It seems true that normal adolescents (Kastenbaum, I959) and also probably younger children structure the future as an al- most Iimitless period and think of death as only a very distant and remote possibility. For the children in our experimental sample, the continual presence of a dying child may necessitate the re-ordering of these time perspectives. The present may also take on unusual meanings if it is bounded by the anticipated 24 death of a brother or sister. Openness of Famiiy Discussion aboUt Illness This part of the study is an extension and partial rep- lication of a finding by Waechter (l968) in a study of 6-IO year old children with fatal illness. She found that children from families judged to be more Open to discussion of illness showed less anxiety than children from families which did not openly discuss the illness. Since the question of how much and what to communicate to ill children and their siblings is of major importance to doctors and other professionals working In this area this appears to be a finding worth con- firming with cystic fibrosis families. Hypotheses To try to answer some of these questions, the following hypotheses were formulated: l. Children with an advanced, terminal case Of cystic fibrosis will express more a) anxiety, b) depression and c) adjustment problems than children who have less advanced cases of cystic fibrosis or who have a chronic, but not fatal, condition. 2. Siblings of children with advanced cystic fibrosis will express more a) anxiety, b) depression and c) will have more adjustment problems than siblings of children who have a less advanced case of cystic fibrosis or who have a chronic, but not fatal, condition. Siblings of children with advanced 25 cystic fibrosis will express more guilt than siblings of children with a less advanced case of cystic fibrosis or those who have a chronic, but not fatal, condition. 3. Children who have an advanced terminal case of cystic fibrosis will have a shorter future time perspective than children with a less advanced case or those with a chronic, non-fatal disease. 4. Siblings of children with an advanced terminal case of cystic fibrosis will have shorter future time perspectives than siblings of children with a less advanced case or those with a chronic, non-fatal disease. 5. Children in the families where communication about illness is fairly Open and direct will experience less anxiety, guilt and depression and adjustment problems than do children in families where communication about the illness is closed and guarded. CHAPT ER TWO METHOD Limitations in Selection of Subjects Studies in the area of psychological research on death and dying are quite severely limited by both practical and ethical considerations. Psychologists are not trusted mem- bers of most hospital or clinic teams. They have only re- cently and In small numbers become Interested in and avail- able to fatally ill patients and the people who treat them. They do not come with a background of medical knowledge. And their methods and their language are foreign to the me- dical staff. Thus, it is not simply a matter of course for the doctor to allow the psychologist access to the families in his charge. For this research I was lucky enough to find some doctors who were willing to set aside a traditional bias against psychologists as interfering and possibly harm- ful outsiders with little or nothing to offer to physicians and their patients.' Another problem is finding in one geographic area a large enough group of children with the same terminal illness to allow for homogeneity in the research subjects. The percen- tage of children in the general population with fatal or po- tentially fatal illnesses is small, and the number who are 1One highly respected doctor to whom a request for subjects was sent proved unable to reply after several months of telephone calls and repeated promises of an answer by se- veral postponed deadlines. 26 27 likely to be treated at any one hospital or clinic is far smaller. This research required access to a major center for pediatric illness. The Children's Hospital Medical Center in Boston is such a hospital. The Children's Hos- pital Chronic Nutrition (or Cystic Fibrosis) Clinic is the largest in the United States with a roster of six hundred patients. We anticipated that the parents of these sick children might be reluctant to cooperate fully with this research, and thus jeOpardize the accuracy and completeness of our data. The parents might be expected to protect themselves and their sick children from unnecessary burdens, especially since the sick children already have a daily regimen of pill-taking, physical therapy, aerosol treatments and diet control. Also, we anticipated that some parents might be uneasy about allowing extended interviews with their children. As will be discussed later, each family works out for itself how much and with what degree of openness it wishes to discuss the presence of cystic fibrosis in the family. The wish to protect the child from knowledge of the consequences and course of the disease be- yond that which the family had already discussed was expected to be a factor in parental refusal to participate. Rather surprisingly, the refusal rate in this study was extremely low and the participating families were most co- operative. Only four families refused to participate yielding a refusal rate of IO per cent. Two refusing parents cited 28 problems of time, one said that they did not want the sick child singled out any more than already necessary, and the fourth cancelled an appointment they had made when problems of divorce, financial pressures including selling their home, and legal problems became too overwhelming. Of the 36 families who did participate many were families where both parents worked and where there was precious little common time for the family to be together. That they were so willing to devote four hours of that time to participate in this project attests, first of all, to the strong positive sense of trust and confidence they had in the clinic doctors and staff. Much of their Openness with me was possible because they saw me as an extension of the clinic staff. The high le- vel of cOOperation also was due to the fact that the doctors had purposely selected families who had a good relationship to the clinic and would be likely to participate in the research. This selection process probably eliminated more disturbed families and families who were having a more difficult time coping with the Illness and who might, therefore, be expected to have a more ambivalent relationship to the clinic. While this preselection lowered the refusal rate and made the inter- viewing easier, it probably also restricts the applicability of the results of this research to families that are fairly well-adjusted and well-adapted to their child's illness. It also seems likely that I was meeting a long-felt need in providing an Opportunity for these parents to discuss both 29 their difficulties and triumphs in adapting to their child- ren's illness. While most of the parents did not feel they had problems of sufficient weight to seek help from the clinic social worker, they were eager when approached and asked to talk about the problems the diaease had caused for them and the solutions they had found. Many talked of how difficult it was to discuss such things with family, friends, or reli- gious figures who were often overcome by the thought of a sick child. I took the position of someone familiar with the medical aspects of CF but eager to know more about how families adapt emotionally to this illness. I often supported the pa- rents in how difficult their particular task was and shared with them similar problems which other parents of sick child- ren had reported.‘ 0n the other hand, I maintained (or tried to) a position of unconditional acceptance and did not make judgments about particular aspects of how they managed their children. In turn, I was rewarded with their remarkably honest and straightforward discussion of family relations, feelings about the illness and its treatment at the hospital, and thoughts and fears about the future. The children, too, were remarkably cooperative. NO child who had begun the rather tedious hour-and-a-half testing session failed to complete it. Again, this reflected the com- mitment and support of the parents who encouraged the young- sters to finish their tests. Several patients and siblings went beyond the requirements of the study in discussing their 30 personal feelings about the Illness and its effect on them and on other family members. Subjects Thirty-six families were interviewed and tested for this research. This included 36 mothers, and testing 36 children identified as the primary patient, and 59 of their siblings. Because both of the diseases studied have at least some hereditary basis, eight families were seen where more than one child had the disease in question. A total of five sib- lings who were also affected were tested, although only two of these were included in the final sample selection.l In each family an attempt was made to test the siblings closest in age to the identified patient. At the conclusion of the research one sibling in each family was chosen to be included in the statistical sample. The selection was made so as to maximize the comparability of groups in terms of sex and rela- tive age of siblings in comparison with their affected brothers and sisters (See Table l for demographic data). The 36 families comprised three groups of twelve each. The main sample (CFI) was composed of l2 families where a child had cystic fibrosis of at least moderately severe inten- sity. It was felt by the clinic doctors that these children were in the terminal or near-terminal stages of their disease, 'The two siblings with cystic fibrosis were included because, in one case, the previously selected healthy sibling could. not participate, and, in the other case, because in the fi- nal selection of siblings, matching of the samples on prime demographic variables, like sex, could be accomplished only by including the affected sibling. 3| Table l Age, sex, age at diagnosis, number of hospitalizations for patients and age, sex and relative age of siblings PatIEnt STinng Disease Family2 Age Sex Age at No. of Sex Age Older Group Name Dx in Hospital- or months izations Younger CF. Abbott 20 M 72 I6 F 8 Younger Barber II F 5.5 4 F l6 Older Crystal I6 M 48 4 M l4 Younger DeCarIo l8 M 78 2 F l2 Younger Friedman II F 30 3 F l9 Older Getz I3 F I IO M 9 Younger Hill 9 F 22 3 M IO OlderI Jefferson l0 F l 3 F l3 Older Larkin I8 F 48 2 F 8 Younger Logan I3 M 3 3 F I7 Older Martin 2i F 4 l6 M l5 Younger White I4 M 6.5 4 M 24 Older CF2 Goldberg l2 M 42 2 M II Younger Greer IS F 69 0 F I9 Older (Hurley 20 M 24 O M l6 Younger ‘Kagan”' l6 F ID I F I4 Younger Kane I2 F 8.5 I M 20 Older McDonald I? M l 4 F -l0 Younger Moore I2 F 2.5 I M l2 Younger' O'Neill 7 M I2 I M IO Older Page II M 24 lo F 9 Younger Peneau 9 F l l M l0 . Older Smith 7 F l I M IO Older Wagner 20 F I92 3 M 9 Younger 08 Austin I3 F I08 9 F I2 Younger Brush l2 M 84 2 M II Younger Cunningham l3 F I32 2 F I9 Older DeMasi l3 M 60 3 F l5 Older Fitzgerald I5 M 48 5 M l4 Younger Howell I2 F 54 3 F l7 Older Jaffe l3 F I20 4 F l5 Older Jones I3 M 54 2 M II Younger Pierce I3 F I20 2 M I2 Younger Sandberg I6 M I68 2 F I8 Older Schwartz l2 F 60 2 M l7 Older Walsh I4 F 60 2 F I8 Older |These siblings also have cystic fibrosis. 2Aii family names are fictitious. 32 and, in fact, five of these children have died as of this writing. The first control or contrast group was composed of I2 families where a child had a mild case of cystic fibrosis (CF2). Despite their illness, these children led relatively normal lives. They attended school, could play quite actively, and did not have the frail, emaciated ap- pearance of the children in the main experimental sample. This group was selected as a comparison sample for the ter- minally ill children in the hope that we could learn from them something about the course of the emotional adaptation of families to cystic fibrosis. One question of particular in- terest was whether it was necessary for the child to be ob- viously physically ill, as in the case of the main sample, for the families to begin to accept and adapt to the child's illness and possible death, or if the knowledge of the diag- nosis, clinic visits and home care of even outwardly healthy- looking children was sufficient to begin the process of adap- tation and preparation for eventual loss. It is known that in families of leukemic children, denial of the illness is quite strong during periods of remission when the child Is not obviously ill (Futterman, I970), and that it often comes as a seemingly unanticipated emotional shock when the symp- toms of the illness resume. In children with cystic fibrosis the period of good health can last for years and I wondered how this affected the family psychologically. All children in these two groups were patients in the 33 Chronic Nutrition (Cystic Fibrosis) Clinic of the Children's Hospital Medical Center in Boston or were private patients of Dr. Harry Shwachman, Director of the Clinic. Designation of severity of illness was made by one of the three senior doctors in the clinic.' Discussion with the Clinic nurse and social workers and a reading of the child's medical chart provided further information on demographic variables, such as presence of siblings, previous deaths from cystic fibrosis in the family, etc. which were necessary to establish eligi- bility for this study and to insure comparability between the groups. The second contrast group was composed of l2 families who had a child with juvenile diabetes. Diabetic children were selected as chronic illness controls to help determine how significant the eventually fatal prognosis was in a fa- mily's psychological adaptation. Like cystic fibrosis,‘ diabetes is a chronic condition necessitating periodic clinic visits and daily home care. It most often runs in families, although the exact genetics of diabetes are not as well under- stood as those for cystic fibrosis. It is a serious illness IAlthough it is possible for doctors to make an objective ra- ting of each CF patient's extent of illness, called the Shwachman rating, the CF Clinic doctors (primarily Dr. Shwachman) who were familiar with the nature of the groups I wanted, preferred simply to designate whether or not a parti- cular child was suitable for one of my two groups. The sig- nificant difference between the two groups in number of hos- pitalizations (mean of 5.8 for the terminally ill group (CF.) and of 2.0 for the mild CF group (CF ) and the fact that five of the I2 children in the first grou have died while none of the children in the CF? group have died is indicative of the accuracy of the doctor 5 selections. 34 that can lead to many severe complications if not properly treated. Juvenile diabetes does not usually carry the fatal prognosis that is part of cystic fibrosis. Juvenile diabetes Is also a reasonably prevalent illness. Therefore, it was felt that it would be an excellent group for assessing the emotional strain on a family of having a chronically, but generally not fatally Ill child and would make a useful con- trast to the terminally ill CF. group. Diabetic children were selected from the population of the Adolescent Diabetic Clinic at Children's Hospital, Boston. Because this adolescent population (from l2 up) was the one available to us, the age range of the children in this group is more limited than those in the cystic fibrosis groups, al- though the average age of the patients in the three groups is not significantly different. Median ages for the CF., CF2, and DB groups are respectively l3, l2, and I3 years. An ef- fort was made to include children whose diabetes was not un- der perfect control and who therefore needed more frequent hospital or clinic visits to modify their regime. The actual selection of subjects frOm the two clinic populations was accomplished in one of two ways. By the first method, the experimenter went through the files of the Cystic Fibrosis Clinic, selecting subjects who met the demographic requirements for eligibility. These requirements were (I) patient is between 7 and 2| years of age; (2) patient has at least one sibling who is between 7 and 2i years of age,‘ IThis requirement was removed in one case where the only available sibling was 24 years Old. 35 (3) sibling lives at home and (preferably) does not have cys- tic fibrosis, (4) there have been no previous deaths from cystic fibrosis in the family, and (5) the family lives In Massachusetts. A list of the names of patients who met these . requirements was compiles and given to Dr. Shwachman. He then decided which of these patients were healthy enough to be In the CF2 group or ill enough to be in the CF' group. He also, however, disqualified patients whose parents would not be likely to cooperate in the research. The reasons for each non-medical disqualification were not discussed at length with the experimenter. The total number of disqualified patients was often one-half to two-thirds of the original list. Since this first method required a great deal of time spent in reviewing clinic records for a small yield in the number Of subjects approved, a second approach was tried. This was to present to Dr. Shwachman a list of the patients scheduled for clinic appointments during the coming week and to ask him to select those who met the medical criterion for inclusion in the CF. or CF2 groups. Dr. Shwachman selected subjects in this way, but he also eliminated patients whose families, he felt, would resent the intrusion and would be unlikely to participate. The experimenter then reviewed the medical charts of the selected patients and chose those who met the demographic criteria. In the Diabetic Clinic the process of selection was sim- llar to the second method described above for the Cystic 36 Fibrosis Clinic. It was the Clinic nurse in the Diabetic Cli- nic who reviewed the lists of patients and selected those who had at least moderately severe diabetes. She also eliminated patients whose families were inconsistent in keeping clinic appointments or were otherwise uncooperative and, therefore, unlikely to participate. Parents Of the selected patients were approached most fre- quently in person during clinic visits or by telephone. In some instances a letten signed by the Cystic Fibrosis Clinic Directors, was sent to the parents explaining the purpose of the research and the nature of the tasks involved (See Appendix A for a copy of this letter). If the family agreed to participate, an appointment was made for the experimenter to visit the home or, in a few instances, for the family to be seen at the hospital during a clinic visit. It must be clear that all three groups were far from ran- domly selected samples. Neither was each subject perfectly matched to a control. To try to do so in the limited number of clinic families available would have been impossible. In- stead, I have tried to remain conscious of the fact that these were, for the most part, highly motivated, conscientious families who sought out the excellent medical facilities of the Children's Hospital Medical Center and who were probably selected by the doctors, either consciously or unconsciously, as families who would be likely to cooperate with our research requests. It seems likely that as a result of the selection 37 biases, the families who were chosen were better-adjusted and better adapted to the illness than the families who were not selected. Beyond the medical criteria, families were selected mostly on the basis of the age and sex of the patients and siblings, with attempts made to keep other factors as equal as possible (e.g. number of children in the family, religion, income, etc.). The three groups are not significantly different in age of the patients or siblings, number of children per family, Income, sex of patients, or relative age of the patient and sib. They do differ in the number of hospitalizations per patient, which is a reflection of the purposely varied severity of ill- ness between the three groups. The difference is in the ex- pected direction with children in the CF| group have a median of three hospitalizations, compared to one for the CF2 group and two for diabetic children. The age at diagnosis was sig- nificantly lower for cystic fibrosis patients than for diabetic patients. The CF. and CF2 groups do not differ significantly in the number of years since diagnosis. Children in the CF. patient group, however, were on the average significantly (p .025) younger at the time of diagnosis than the diabetic patients. This is a function of the diseases themselves. Cystic fibrosis is a genetic disorder which can be diagnosed at birth, while juvenile diabetes cannot be diagnosed until symptoms appear in childhood or adolescence (See Table l and Appendix B for a listing of demographic variables.) 38 The families were seen, for the most part, in their homes which were within a 60 mile radius of Boston. Six families were seen at Children's Hospital during a period when their child was an in-patient. Three families were seen on an out— patient basis at the hospital, usually on the day of a clinic visit. One subject was interviewed at her college dormitory. Procedure In brief, measures of anxiety, future time perspective, adjustment, depression, guilt, and familial inter-relationships were obtained from CF., CF and DB patients and at least one 2! of their siblings. Mothers of the patients were interviewed to obtain information on the family's adaptation to the ill- ness and demographic data. I was introduced to the family as a psychologist from the Children's Hospital Cystic Fibrosis Clinic (with the diabetic group, as from the Adolescent Diabetic Clinic) who wanted to know more about the families served by the clinic. The family was told that they had been selected not because of any parti- cular dimension of their child's illness, but because they had met some of the demographic criteria of the research, such as having more than one child between the ages of 7 and 2i, living within a 60 mile radius of Boston, etc. It should be noted that hearing this was particularly reassuring to the sick patients who seemed to have assumed that their inclusion in the research group either meant that they had a particularly 39 serious medical case, or that the doctors felt there was some- thing psychologically wrong in their dealing with the illness. The procedure most often followed was to interview the mother while the patient and sibling(s) worked on the three written, self-administering tests - the California Test of Personality, the General Anxiety Scale for Children, and the Incomplete Sentences Test. The children were given instruc— tions for these three tests before the maternal interview was begun and were told that they should feel free to return if they had any later questions about the tests. The children were asked to go into a room which was as separate and distant as possible from the room where the maternal interview was held. It was explained that it is difficult for children to concentrate on their tests if they can overhear a conversation in which their mother is talking about them or about other ' The children were asked not to communicate family members. with each other about the answers to the tests at the time of the testing, although they were free to discuss their answers if they wished to at the completion of the testing. |In a few families, such separation was not physically pos- sible without sending the children to their rooms. In one such family which was not amenable to this idea, the maternal interview was carried on in a room separated only by an open archway from the room in which the children were doing the written tests. This situation provided an interesting example of the distortion of communication which can exist in a family where one member is terminally Ill. Although only a few minutes earlier the sick child had called out from the adjoining room to correct something her mother had said about her friend- ships, the mother told me in an equally loud voice that this little girl did not know about her expected limited life span and that she and her husband meant to spare the child this knowledge. 40 The maternal Interview and the written tests usually took approximately the same length of time, an hour to an hour and a half. At the conclusion of the interview, the mother was requested to leave the room and one child was asked to join the examiner who then administered the projec- tive tests and the Family Relations Test. Subsequently, each of the other participating children was seen individually to complete the projective testing and the Family Relations Test. If any of the subjects were too young to read, the three written tests were read to them and the answers recorded by the examiner if necessary. All tests were presented to the subjects in the same or- der except for occasional variation due to pressures on sub- jects' time. The order of presentation was as follows: (I) California Test of Personality, (2) General Anxiety Scale for Children. (3) Incomplete Sentences Test, (4) Thematic Apper- ception Test, (5) Waechter Projective Pictures, and (6) Family Relations Test. This standard order of presentation minimized the possible contamination effects that might have occurred, for example, if a terminally ill child was given first the In- complete Sentence Test requiring him to think about his future and then was tested for his level of anxiety. galifornia_1est of Personality. The I953 revised edition of the California Test of Personality was administered to all patients and siblings to obtain a measure of overall psycho- logical functioning. The Total Adjustment score, a composite 4| score based on l2 subtest scores, was used as a general measure of personal and social adjustment. Scores on several subtests, thought to be of particular relevance to the sample studied in this research, were also examined. These included the sub- tests on Self-Reliance, Sense of Personal Worth, Withdrawing Tendencies, Nervous Symptoms, and Family Relations. Since sub- jects In this research ranged in age from 7 to 24, it was ne- cessary to use the Elementary, Intermediate, Secondary, and Adult forms of the test. For comparison of personal adjust- ment scores across forms, percentile scores from the four forms were converted to standard scores. The test, which is composed of l80 items at the upper levels and I44 items at the elemen- tary level, is designed to measure aspects of internal security, and also of social skills and competence. Validity studies have indicated that the CTP is useful in eliciting a persona- lity profile similar to that obtained in a controlled inter- view (Thorpe, Clarke, and Tiegs, I953). General Anxiety Scale for Children. The anxiety scale used in this research was the General Anxiety Scale for Child- ren (GASC) developed by Sarason et al. (l960). Originally, the Test Anxiety Scale for Children, deveIOped by the same group, was also given to the subjects in this research, but it was found to be inapproOriate for subjects who had finished school or who were too sick to attend school because the items referred toospecifically to present functioning in the class- room. It's use was therefore discontinued and the majority of 42 subjects received only the GASC. The GASC is a 45-item scale that measures the subject's reaction to common sources of anxiety. The subject Is re- quired to answer “Yes" or "No” to questions, such as, ”Are you afraid of spiders?" (See Appendix C). The questions elicit feelings about separation, mutilation, fear of III- ness in oneself and other family members, phobic behaviors, and anxiety in school or other achievement situations. The subject's score is the number of items to which he answers "Yes," i.e. admits having felt anxious under the conditions described. While the Sarason group was aware that the for- mat of the test left Open the possibility that some high scores would be the result of acquiescence rather than high anxiety, they felt it necessary to keep the simple format to enable first and second grade youngsters to take the test. They also did further work that failed to demon- strate a significant relationship between acquiescent ten- dency in children and high anxiety scores on the GASC. A more serious issue, and perhaps a more relevant one in this research, is the question of defensiveness, or the ten- dency of subjects to lie about anxieties which they do, in fact, experience. The GASC contains an ll-point lie scale, composed of statements about anxieties which nearly everyone experiences. It is felt that if a subject answers "No" to such questions, thereby denying that he experiences these common anxieties, then he is likely to be lying about his anxieties 43 on the whole scale to a degree which might be likely to sig- nificantly alter his overall score. Sarason et al. (I960) found correlations of -.40 to -.66 between lie scores (i.e. number of lie items answered ”No”) and GASC scores. They also discussed the tentative finding that a tendency not to admit to experiencing anxiety was related to a more general tendency not to admit to unfavorable traits about oneself. Because this defensiveness about admitting to the experience of anxiety seemed to be an important factor in evaluating GASC scores and one which might well be subject to situational variation, such as the presence of illness In the family, the lie scores of subjects In this research were also calculated and analyzed. The GASC is a widely used anxiety scale on which many care- ful validational studies using large sample Of children and adolescents have been done (Sarason et al., I960; Cowen, I957). Incomplete Sentences Test. The Leasing Incomplete Sen- tences Test (Leasing, l972) was used as a measure of future time perspective. This nine-item test is composed of sentences such as, ”I often think of what I will do within the next years,“ or 'I don't see much sense in planning more than years ahead,” (See Appendix 0). The subject is required to complete each sentence and the score is the median number of years of future extension reported. Leasing believes this test to be a measure of cognitive future time perspective which she defines as, "the time span over which personal 44 future images are projected" (I972). This test is particue larly suitable to a population covering a wide age range, as in this research, since the questions are not defined by par- ticular life events which might be highly influenced by the age of the subjects. Rather, it is a fairly direct measure of the subjects' extension of personal time In a free fantasy situation which seemed appropriate for both the youngest (age 7) and the oldest (age 24) subjects in this research. Leasing (I972) reports normative data for I68 girls, ages 9-l5, on this version of the IST and also reports data on an earlier version of the test from a study of 746 boys and girls in Grades 5, 8, and II (l968). Projective Testing. A set of eight pictures was shown in- dividually to each patient and sibling in this research. The subjects were requested to use their imagination to tell a story about each picture. They were asked to tell (I) what was happening in the picture, (2) how the peeple in the picture felt, and (3) how the story would end or what would happen next. The set of eight pictures was composed of four cards se- lected from the Thematic Apperception Test and four cards which were designed for and used by Waechter (l968). It was hoped that these pictures would elicit feelings and attitudes more directly related to the child's Illness than the TAT cards. A list and description of the pictures used is presented in Table 2. The four TAT cards were presented first to Introduce the child to the nature of the task and to elicit more generalized 45 TABLE 2 Projective Test Pictures Order Description Source Given I. Small child sitting in doorway of cabin TAT l3 8 2. Woman entering rOom, face on hand TAT 3 GF 3. Figure outlined In open window TAT I4 4. Boy in front of mural depicting operation TAT 8 BM 5. Two boys in adjoining beds Waechter 6. Small child in hallway outside closed Waechter door to Intensive Care Unit 7. Child in bed, parents and doctor out- Waechter side door 8. Small child in bed, nurse nearby with back turned Waechter fantasy before introducing the pictures of hospital situations. The Waechter cards were then presented in the order listed. Card I of two boys in adjoining beds was given first. This card, which presents the fewest hospital cues of the four, was seen by some children as taking place in a home rather than a hospital. The picture of a little girl outside a door marked "Intensive Care Unit" was given next. For younger children an explanation was given of an intensive care unit as a place in a hospital for very sick peOpIe who require a lot of special care from the doctors and nurses. Card 3, a picture of a girl lying in bed with eyes closed and three adults, presumably the parents and doctor, talking just outside her door, elicited the most 46 fantasy about severe illness and death. Card 4, a more benign picture of a small child in bed with a nurse nearby, was given last. The cards were presented to each child In as nearly pri- vate a setting as was possible within the home or hospital. Other family members were requested to leave the room If present so as to allow the person being tested to use his imagination more freely. Siblings who were also participants in this re- search were, Of course, asked to stay out of the room when their brother or sister was telling his or her stories. Despite the young age of some Of the subjects, all were able to complete the task Of telling the full set of eight stories. No time limits were given. The experimenter did remind subjects of the original questions about affect or outcome if they failed to include them in their story. The stories told by the subjects were transcribed to typed sheets from the written recordings taken by the experimenter. These were then rated for depression and guilt by an independent judge for whom reliabilities of r-.87 for depression and r=.9l for guilt had previously been established. The rater was an ex- perienced psychological tester who was familiar with projective techniques, but naive of the hypotheses of this research or of any Information about the subjects. The transcribed stories were identified by numbers only. The experimenter and the judge had established reliabilities by making independent ratings of both guilt and depression from over IOO projective stories. The level 47 of agreement between the experimenter's ratings and the rater's ratings was calculated using the Pearson product-moment formula. The 5-point rating scales for Depression and Guilt were de- vised by this author and are presented In Appendices F and G. The depression scale ranges from happy, positive effect through mild depression to severely depressed or suicidal feelings. It is based on the description of the main character's affect as given by the subject. The guilt scale attempts to measure the degree to which the main character in the story feels responsible or remorseful for any negative consequences of his own behavior. {smily Relations Test. The Family Relations Test (Bene and Anthony, I957) Is an instrument that assesses the direction and intensity of the child's feelings towards the members of his family and his estimate of their reciprocal feelings for him. It was used in this research to obtain a measure of the degree to which each child felt he or his siblings were overprotected and overindulged by their parents and also to measure the inten- sity of negative feelings felt for the sibling who was the partner in this research. The test materials consist of 20 figures representing people of all ages from grandparents to babies and a set of 86 cards each containing a statement about the expression of a particular affect within the family (for example: "Some- times I hate this person in the family,“ or "This person in the family cares for me more than for anybody else.”) The clld was then asked to read each card silently and to place it in 48 the box of the person in the family for whom the statement was true. If it was not true for anyone in the family, the card was to be placed in the ”Nobody” box. If it was true for more than one person in the family, the subject gave the card to the experimenter and told her which people In the family it applied to. All subjects in this research were given the form for Older children (i.e. over six years of age). Subjects who could not read had the statements read to them by the experi- menter, but then they themselves filed the card in the approp- riate box. All subjects seemed quickly to understand the task. Of all the tests given for this study, this was by far the subjects' favorite. Many seemed very emotionally involved in the card-sorting, taking great pains in deciding which family member answered the description on the card. Several children expressed their involvement with the task by teasing parents or sibs afterwards about the things they had ”said” about them during this test. Scoring was accomplished by use of a grid which broke down the affects expressed Into categories of incoming and outgoing, strong and mild, negative and positive emotions. It also con- tained sections for recording the expression of feelings related to parental overindulgence and overprotection. Within each category, spaces were provided for recording the number of times an affect within that group was attributed to each family mem- ber. A listing of the items which made up the Overprotection 49 and Overindulgence and Negative Affect categories is listed in Appendices H and I. In this research the relevant scores for the measurement of Overprotection were (I) the number of times the subject indicated that his parents overprotected or overindulged him, and (2) the number of times a subject indi- cated that his parents overprotected or overindulged the sib- ling who was also participating in this research. A composite score, hereafter referred to as the "Self-Sib Overprotection Score” was obtained by subtracting the sib score from the self score (No. 2 above from No. I). The relevant score for measurement of Negative Affect Towards Sibling was the number of times a subject expressed mild or strong negative feelings towards the other participating sibling. Bene and Anthony (I957) report high levels of agreement between Family Relations Test scores and predictions based on clinical material for children in treatment at a psychiatric clinic. Frost (I969) found that the distribution of affect within the family, as measured by the Family Relations Test, was markedly different for delinquent and non-reader ll-year- old children than for normals of the same age. Although this test has not been widely used and researched, the enthusiastic and intense response that it generated in the subjects en- couraged the experimenter to believe it is a valid measure of the children's feelings about their family members. Maternal Interview. An interview was conducted with the mother of every patient in this study. All but eight of the 50 mothers were interviewed in their homes. The other eight were seen at Children's Hospital during an out-patient clinic visit or during a period when their child was hospitalized. The interview took one to one and a half hours and was usually accomplished while the children were doing the written part of their tests. The interview schedule used In this research was, with a few minor changes, the one Used by Waechter (I968) in her study of fatally ill children. The earlier sections of the interview elicited Information on the child's early develop- mental history, his school history, his relations with peers and siblings, and the nature of the parent-child relationships. The second section was devoted primarily to questions about the illness and its effects on the family. In both sections, questions were asked so as to minimize any threat to the mother and to reduce the tendency for the mother to answer in a so- cially desirable way to please the experimenter. Some ques- tions were short and factual, while others asked for the mother's Opinion on Opposing approaches to child-rearing in particular situations. In the latter case, each side was sup- ported equally by the examiner in the hope of getting the mother to feel comfortable in expressing her own true views on the subject. The purpose of the interview was threefold: (I) to pro- vide specific demographic information about the family, (2) to evoke a general picture of the family's adaptation to the 5| illness, and (3) to elicit particular information about variables felt to be central to these adaptive processes. These included information on (I) the openness of the family in discussing the illness, (2) the warmth of the mother-child relationship, (3) the mother's affect level, (4) her judgment of her sick child's affective state, (5) the level of burden experienced by the mother as caused by the illness, and (6) her estimate of the sick child's needs for discipline relative to the needs of his siblings. With the exception of the mother's affective level and the warmth of the mother-child relationship which were evaluated by a more global judgment of the whole Interview, , the variables were judged primarily on responses to particular questions within the schedule. The judgment was made by an in- dependent rater with whom reliability of r=.97 had previously been established. Rating scales for the six variables were deveIOped to code the information. The rating scales for Openness in Discussing the Illness and Warmth of Mother-Child Relationship were modi- fied from scales used by Waechter in her research. The other four scales were deveIOped by this experimenter. The rating scales are reproduced in Appendices K-P. In addition, the maternal interviews provided information on early developmental difficulties experienced by the Patients often as a result of their disease, and on the later psycholo- gical symptoms which the physically ill child deveIOped. Because the physical symptoms of cystic fibrosis most often 52 become apparent before age five, and can be apparent at birth, there is, in most cases, little time for the deveIOp-. ment Of a normal mother-child relationship prior to the on- set of the child's illness. The most frequent symptom that drives the mother to seek a doctor's care for her child is the child's failure to gain weight. While the children often eat continually they seem to be constantly hungry, have an excessive number of bowel movements, and do not gain weight. Many mothers, especially those who had not had other children, began to blame themselves for their baby's failure to thrive. Further problems developed if the disease had not been diagnosed by the time the toilet training started since bowel. control is more difficult to achieve when movements occur so frequently and when they are a source of such special concern for the parent. In addition, the particularly foul-smelling stools that are symptomatic of cystic fibrosis can add to the mother's displeasure at the child's inability to care for himself in this area. At the time when many of the children in this study were age five and under (and even today), relatively few doctors outside of CF centers are experienced enough with the symptoms of CF to make an early diagnosis. Many of the mothers in this study reported a long, frustrating period, sometimes several years In duration and Involving several hospitalizations, be- tween the time they first noticed symptoms and brought the child to a doctor and the time the diagnosis was made. The 53 severity of the frustration was reflected in the fact that several mothers reported feeling ”relieved” at the time of the diagnosis. Despite their awareness of the serious nature of the illness, the mother's knowledge that the child's ill- ness had finally been identified, that appropriate treatment would soon begin, and that their direct responsibility for the child's failure to thrive had been disproven led to a feeling of relief for these mothers. Since It seemed important to try to assess the impact of these early disturbances in the mother-child relationship, Early Developmental Difficulties, including feeding, sleeping, and toileting problems, hospitalization before age five, and mother's expression of marked insecurity about the child's failure to grow properly were recorded and correlated with other data. The list and description of the Early DeveIOpmen- tal Difficulties is given in Appendix Q. The maternal interview also provided information which made possible a listing of the recent emotional difficulties experienced by each patient as reported by his mother. These Psychological Symptoms included trouble making friends, bed- wetting, academic failure or major vacillation of grades, fears, nervousness requiring tranquilizers, etc. A full list of the Psychological Symptoms appears in Appendix R. Because the interview schedule was less directly aimed at eliciting in- formation about siblings, it was not possible to compile an accurate listing of the Psychological Symptoms of the siblings 54 as reported by their mothers. StatisticalAnalysig The data which resulted from the tests given to patients and siblings were analyzed using a 3 (Patient Group) x 2 (Sex of Patient) x 2 (Relative Age Of Patient and Sibling) ANOVA with repeated measures. The data on ratings taken from ma- ternal interviews were analyzed using single one-wayiANOVAs. Further exploration of the Interactions found to be signifi- cant on ANOVA was performed through analyses Of Simple Effects (ASE); and analyses of differences among the patient groups were done using Dunnett's Test. Product-moment correlations were used to compare two sets of scores. Occasionally, simple t-tests were employed to explore hypotheses which were not specifically tested by the ANOVA or ASE. The one-tailed .05 .3 confidence interval was taken as the level of significance. Results within the\.lgggfititailed confidence interval are reported as 'approachIng significance.“ The analyses were performed within a framework Of experimental design based on Winer (l97l). CHAPTER III RESULTS Section A: Original Hypotheses For the most part, the original hypotheses put forth in this research were not supported. First, findings relevant to the original hypotheses are presented. Analysis of re- lated data is presented in the following section (Section B). Tables 3, 4, and 5 present cell means and Table 6 presents a summary of the statistical analyses done to test these hypotheses. Anxiety Children with advanced, terminal cases of cystic fibrosis did not show significantly more anxiety than children with less advanced cystic fibrosis or with diabetes, as had been pre- dicted. Contrary to the hypothesis, no significant differences in anxiety levels between disease groups were found. The hypothesis that CF. siblings would be more anxious than CF2 or 08 siblings was not supported. There were no sig- nificant differences in anxiety levels between siblings of children with terminal cystic fibrosis and siblings of child- ren with mild cystic fibrosis or diabetes. When the anxiety scores on the GASC were broken down for males and females, some sex differences were found. These data are discussed further in the section on related findings. 55 56 Table 3 Table of means and standard deviations for patients' scores on the GASC, Depression and Guilt ratings, Total Adjustment scores of the California Test of Personality, the Lessing Incomplete Sentences Test, GASC Lie Scale, SELF-SIB Overprotection score, Negative Affect to Sibling, Early DeveIOpmental Disturbances, and Number of Psychological Symptoms Tests Group GASC Depression Guilt Total Incomp. Rating Rating Adj. Senten. Mean 8.0. Mean 3.0. Mean 8.0. Mean 8.0. Mean S.D. CF| l7.42 7.75 3.08 .39 l.25 - 44.l 8.07 7.89 5.57 CF2 l3.83 7.42 3.20 .56 l.26 - 46.5 7.65 Il.50 5.96 DB l2.75 6.36 2.92 .37 l.l5 - 4I.42 I3.3l 6.63 2.l2 Lie Scale SELF-SIB Negative Early Dev. Psychol. Overprot. Affect Disturb. Symptoms Mean 8.0. Mean 8.0. Mean 8.0. Mean 8.0. Mean 8.0. CFI 2o42 203' 3.63 30'4 2o09 " 200 lei} 3067 "' CF2 3.I7 l.85 0 5.20 3.92 - 2.42 l.3l 2.50 - DB 2.83 l.99 l.36 2.25 2.50 - .67 .49 3.08 - 57 Table 4 Table Of means and standard deviations for siblings' scores on the GASC, Depression and Guilt ratings, Total Adjustment scores of the California Test of Personality, the Leasing Incomplete Sentences Test, GASC Lie Scale, SELF-SIB Overprotection score, Negative Affect to Sibling, Early Developmental Disturbances, and Number of Psychological Symptoms Tests Group GASC Depression Guilt Total Incomp. Rating Rating Adj. Senten. Mean 3.0. Mean 8.0. Mean 8.0. Mean 8.0. Mean S.D. CF. I6.58 6.50 3.I0 .55 l.24 - 42.l 6.48 Il.50 Il.50 CF2 ll.75 7.33 3.I3 .47 l.l5 - 43.5 8.07 9.00 8.04 DB I2.58 7.20 2.89 .30 l.09 - 44.3 9.l0 5.25 3.25 Lie Scale SELF-SIB Negative Early Dev. Psychol. Overprot. Affect Disturb. Symptoms Mean 8.0. Mean 8.0. Mean 8.0. Mean 8.0. Mean S.D. CF. .075 .005 -3025 5.66 '092 "' "' " - "' CF2 3083 2037 “.075 2056 5083 " " - " " DB 2042 I056 -2000 2059 2083 - - - " '- 58 Depression The hypotheses that terminally ill cystic fibrosis pa- tients and their siblings would be more depressed than chronically lll CF or DB patients and their siblings were not substantiated. Analysis of the data showed no signifi- cant differences in levels of depression between groups of patients or siblings. Patients and siblings in all groups appeared, on the average, to be mildly depressed. Average scores for all groups were around 3 (mild depression). Depression levels for cystic fibrosis patients and siblings were slightly higher than for diabetic patient and sibling groups. The expectation that family members would be more de- pressed In families where a child is terminally ill than in families where a child is chronically III was upheld in the findings on mothers' depression levels. CF. mothers expressed significantly more depression than diabetic mothers (p4 .005) and nearly significantly more depression than the CF2 (p< .IO) (See Table 5 for the cell means for maternal variables.) Yet, on the average, none of the groups of mothers was very de- pressed. CF. mothers, the most depressed group, had an average depression score of 2.64 on a S-polnt affect scale where 3 equals mild depression. 59 Table 5 Mean Ratings of Maternal Openness to Discuss Illness, Warmth, Level of Burden, Judgment of Sick Child's Mood, and Sick Child's Need for Discipline taken from Maternal Interview Maternal Variable CF. CF2 DB Openness to Discuss Illness 3.92 3.75 4.64* I Warmth to Sick Child l.67 2.25 l.72 Mother's Depression 2.50 l.75* l.25*** Level of Burden Caused by Illness 3.42 2.83 2.64* Mother's Judgment of Sick Child's Mood 3.08 l.67** 2.27* Sick Child's Need for Discipline Relative to Siblings 2.50 3.08 2.9l ILevels of significance reflect differences in comparison to CF. means. “”9 4 .005 **p < .025 *p¢:.l0 Adjustment The hypotheses suggesting significantly more adjustment problems in CF. patients and their siblings were not upheld on the basis of the Total Adjustment scores on the California Test of Personality. No significant differences between groups were found. The hypothesis that CFI patients would have more adjust- ment problems than CF or DB patients was further analyzed by 2 examining the number of psychological symptoms reported by the F scores from 3 x 2 x 2 ANOVA with repeated measures 60 Table 6 on patients and siblings for anxiety, depression, total adjustment scores on California Test of Personality, guilt, and future time perspective scores on Incomplete Sentences Test df Anxiety Depres- Total Guilt FTP __» sion Adj. BETWEEN * A (Disease group) 2 4|.OO