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High Utilizing Primary Care Patients with
Medically Unexplained Symptoms

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Francesca Colecraft Dwamena

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HIGH UTILIZING PRIMARY CARE PATIENTS WITH MEDICALLY
‘UNEXPLAINED SYMPTOMS

By

Francesca Colecraﬁ Dwamena

A THESIS

Submitted to
Michigan State University
In partial fulﬁllment of the requirements
for the degree of

MASTER OF SCIENCE
Epidemiology

2007

ABSTRACT

HIGH UTILIZING PRIMARY CARE PATIENTS WITH MEDICALLY
UNEXPLAINED SYMPTOMS

By

Francesca Colecraﬁ Dwamena

Some patients with symptoms have diseases but for 50% or more of ambulatory patients,
and 7 -9% of hospitalized patients, either no organic disease exists, or their diseases do
not explain their symptoms. Many of these patients with medically unexplained
symptoms (MUS) become high utilizers of healthcare resources without getting any
relief. Both they and their physicians become progressively ﬁ'ustrated because until
recently, there had been little treatment for them. This thesis summarizes advances in the

ﬁeld over the last 10 years.

In the ﬁrst chapter, I provide a critical review of the epidemiology, diagnosis, and
treatment of MUS. In chapter 2, I explore potential mechanisms of MUS with a
systematic review of neuroradiological studies. The ﬁnal chapter is a qualitative study of
typical high utilizing primary care patients with MUS. This unique study of the
experiences, perceptions, and behaviors of MUS patients has uncovered important

questions for future quantitative studies.

To My Parents, Emmanuel and Victoria, and to my soul mate, Ben, who have always
been my unmovable rock

iii

ACKNOWLEDGEMENTS

I would like to thank my mentor, Dr. Robert Smith for his guidance, encouragement, and
faith. You have taught me so much in so many ways. Your dedication, scholarship, and

generosity are truly inspiring.

I thank Dr. Joseph Gardiner and Dr. Brian Mavis, my committee members, for their

patience, support, and feedback.

Much of this work was funded by the National Institute of Mental Health grant
MH57099-02. I thank Blue Cross Network of Michigan for their active collaboration
with the parent project, Ann Marie Hodges for her help with recruiting patients and Ting
Hong for her help with transcription. I also thank reviewers of earlier versions for their
critical comments. This qualitative paper was presented in part at the Society of General

Internal Medicine annual meeting in May 2004.

Special thanks to Christina Bott. I am truly blessed to have you as my assistant.

iv

TABLE OF CONTENTS
LIST OF TABLES ................................................................................................. vi
LIST OF ABBREVIATIONS ............................................................................... vii
CHAPTER ONE ...................................................................................................... 1
CHAPTER TWO ................................................................................................... 25
CHAPTER THREE ............................................................................................... 37
APPENDIX 1 ......................................................................................................... 58
REFERENCES ...................................................................................................... 60

LIST OF TABLES

Table 1 Prevalence of Lifetime Anxiety and Depression in High

Utilizing Patients with MUS ................................................................. 14
Table 2 Summary of Studies on Sexual and Physical Abuse ............................ 15
Table 3 Systematic Review - Study Demographics and Quality ...................... 33

Table 4 Systematic Review - Summary of Studies and Extracted Variables ....34
Table 5 Qualitative Study - Preliminary and Working Themes ........................ 55

Table 6 Qualitative Study - Final themes with Deﬁnitions .............................. 56

vi

LIST OF ABBREVIATIONS

MUS ............................................................................. Medically Unexplained Symptoms
DSM ................................................................................ Disorders Somatoforrn Disorders
SD .................................................................................................... Somatization Disorder
IBS ............................................................................................... Irritable Bowel Syndrome
SSRIs ..................................................................... Selective Serotonin Reuptake Inhibitors
TCAs ............................................................................................. Tricyclic antidepressants
SCL-90-R .................................................................... . revised Symptom Checklist 90
DIS ..................................................................................... Diagnostic Interview Schedule
NURS .............................................. Naming, Understanding, Respecting, and Supporting
RCT ....................................................................................... Randomized Controlled Trial
MAI ....................................................................................................... Minor Acute Illness
MCS ..................................................................................... Mental Component Summary
CBT ................................................................................... Cognitive Behavioral Treatment
OR ........................................................................................................................ Odds ratio

vii

CHAPTER ONE

Epidemiology, Diagnosis, and Treatment of Medically Unexplained Symptoms — A
Critical Review

Francesca C. Dwamena, MD

1. Introduction

In spite of costly diagnostic testing each year, about 33% of the 400-million
physical complaints in ambulatory patients can be classiﬁed as medically unexplained
symptoms (MUS) [1-4]. Patients with medically unexplained symptoms (MUS) account
for at least 50% of outpatient visits, and almost 10% of inpatient admissions. Either these
unfortunate patients have no organic disease or, when they do have organic disease, the
disease does not explain the presence or severity of their symptoms [5, 6]. Most patients
with MUS have primary psychological problems that are overlooked by physicians’
unending but futile attempts to ﬁnd an organic disease explanation [7-11]. Attended by
worry, these patients become high utilizers of healthcare resources. Unnecessary
diagnostic tests [12-15], hospitalizations [15-17], and surgery [15, 18-21] in these patients
lead to high iatrogenic complication rates and excessive utilization [14, 17, 21]. The
physician becomes progressively more frustrated with his/her failure to ﬁnd organic
disease [12, 22-24] and often conveys wittingly or unwittingly that the problem is “all in
the head” of the patient. The unhappy patient then leaves the physician, ﬁnds a new one,
and the process begins again [12, 22-24].

Although primary care physicians are familiar with MUS patients, the spectrum of
MUS in primary care had not been elucidated as most studies of MUS have focused on a

small subset of patients.

2. Deﬁnition and classiﬁcation of MUS in the Literature
2.] Literature review
According to the Diagnostic and Statistical Manual of Mental Disorders,

medically unexplained symptoms (MUS) are physical symptoms that suggest, but are not

fully explained by, a general medical condition; or the direct effects of a substance or by
another mental disorder. This deﬁnition requires the exclusion of organic and mental
disease, yet studies show a higher prevalence of symptoms without pathology in patients
with chronic medical conditions independent of the severity of their medical disease [25].
Thus this deﬁnition may hinder the study of unexplained symptoms in patients with
chronic organic disease. Lipowski deﬁnes somatization as “a tendency to experience and
communicate somatic distress and symptoms unaccounted for by pathological ﬁndings,
to attribute them to physical illness and to seek medical help for them.” This deﬁnition
does not exclude comorbid organic disease and highlights the importance of the patient’s
health seeking behavior. Many patients with MUS never or seldom seek care for it [26-
28]. The problem for healthcare arises, when MUS leads to excessive utilization, or when
it complicates the presentation and management of patients with organic or mental
disease.

Classiﬁcation of MUS has been difficult. Medical specialties have described
several functional somatic syndromes such as ﬁbromyalgia, irritable bowel syndrome
(IBS) and the chronic fatigue syndrome [26]. Although controlled studies have suggested
the presence of biochemical and physiological abnormalities in many patients with these
disorders [29, 30], none of the disorders can be deﬁned with speciﬁc tissue pathological
changes [31]. Each of the syndromes is seen in a heterogeneous group of patients, has a
higher than expected association with psychological disorders like anxiety and depression
and is often refractory to standard medical treatment [32, 33]. Furthermore, the high
overlap and co-occurrence [34-36] seen in this disorder has called the notion that they are

distinct entities into question and many believe that they should instead be conceptualized

as different manifestations of the same problem [32, 33, 37]. The Diagnostic and
Statistical Manual of mental Disorders Somatoform Disorders (DSM) provides the only
comprehensive classiﬁcation of medically unexplained symptoms to date. Of the seven
disorders identiﬁed (somatization disorder, undifferentiated somatoform disorder,
somatoform disorder not otherwise speciﬁed, conversion disorder, pain disorder, body
dysmorphic disorder, and hypochondriasis.), only somatization disorder has been
validated [3 8]. The fourth edition of DSM (DSM-IV) deﬁnes somatization disorder based
on the patient’s age at onset (before 30 years), chronicity (more than 6 months), number
of symptoms (8 of 35 speciﬁed symptoms), and absence of organic disease. Somatization
disorder is very rare with an estimated prevalence of 0.06 — 1.4% in primary care,
presumably due to its stringent criteria [39, 40]. However, even the residual category,
“somatoform disorder not otherwise speciﬁed”, which requires at least one physical
complaint, lasting more than 6 months and causing signiﬁcant distress, ignores all
chronic symptoms not included in the pre-speciﬁed list of 41 symptoms as well as
recurrent or transient symptoms.

In a chart review of 883 patients with 6 or more visits in the previous year, Smith
et al.. found that 51% of patients had multiple visits for different, minor transient
symptoms, typically with little or no investigation [41]. This high utilization for multiple
minor problems persisted over 2 years. A more recent study that used a reliable chart
rating method as the gold standard [42] demonstrated that all four DSM-IV speciﬁc
somatoform categories (somatization disorder [SD], hypochondriasis, conversion, and
pain disorder) combined with the less restrictive ‘abridged SD’ [43] comprised less than

75% of all high utilizing primary care patients with MUS [44]. Thus, the majority of

primary care patients with MUS may not ﬁt any DSM category. Yet these patients may
have signiﬁcant functional and psychological disability because of excessive worry.
Conelly et al. [45] found that 21% of primary care patients had poorer health perception
scores than expected for their level of physical health as rated by their physicians. These
patients felt to be physically healthy by their physicians had more health worry, acute

pain and depression than those with higher health perception scores.

2.2 An Alternative Classification System for Medically Unexplained Symptoms

We have proposed an alternative classiﬁcation system that conceptualizes patients
with MUS as a heterogeneous group with a spectrum of presentations ranging from mild
to severe [46, 47] based on the number and duration of symptoms they experience and
amount of healthcare they utilize [47]. The individual symptoms can be of any intensity
and comorbid organic and psychiatric diseases are common across the entire severity
spectrum of MUS. Psychological dysfunction, functional disability, and prescription and
non-prescription substance misuse [48, 49] increase with increasing severity of MUS.
Patients with severe MUS are also more likely to have a history of physical and sexual

abuse [50, 51].

2.2.1 Mild MUS (about 80% of all MUS patients in clinical settings [47]

Patients with mild MUS experience unexplained symptoms, usually one or two in
number, which last from days to weeks and do not lead to high healthcare utilization [2,
52]. Although these patients often undergo unfruitful diagnostic tests [2], their condition

is usually not recognized as MUS because the symptoms are self-limited and usually

present little difﬁculty for clinicians. Nevertheless, making mild MUS explicit as a
diagnosis may help resolve the problem of excessive laboratory testing, unnecessary
treatments, and iatrogenic complications. These patients have not been studied [53] and
are often considered “noise in the system” that is difﬁcult to differentiate from normality
[46, 47]. Psychosocial factors in patients with mild MUS are assumed (e. g., “stress” and

worry) [54], but have not been speciﬁcally studied.

2.2.2 Moderate MUS (about 15% of all M US patients in clinical settings [ 4 7] )

Patients with moderate MUS have unexplained symptoms that last from one to six
months and high rates of healthcare utilization (e.g., they have an average of 8 or more
clinic visits a year) because of multiple recurrences of the same or different sets of
symptoms (i.e., those with minor acute illness) [41]. These “worried” [45] patients often
present a signiﬁcant challenge for clinicians presumably because of psychological
dysfunction which is difﬁcult to diagnose and treat as they often do not meet criteria for
any deﬁned psychiatric MUS entities [42]. Prevalence of psychiatric disorders including
depression, anxiety, dysthymia and other psychiatric problems is estimated at 20% in

patients with moderate MUS [47].

2.2.3 Severe MUS (about 5% of all M US patients in clinical settings [4 7])

Patients with severe MUS have chronic, persistent and multiple unexplained
physical symptoms of at least 6 months duration that result in signiﬁcant functional
impairment and excessive utilization of healthcare resources [55]. These patients are well

recognized and perceived as “difﬁcult” by primary care clinicians, who lack conﬁdence

in their ability to care for them [22, 56]. Subsets of severe MUS have been studied
extensively under entities whose validities have been questioned because of signiﬁcant
overlap in symptom criteria [6, 32, 46]. These entities include Somatoform Disorders [5]
in the psychiatric literature and Functional Somatic Syndromes [32, 46] in the medical
literature. Studies suggest that at least 67% of patients with severe MUS meet criteria for

some psychiatric disorder [43] and that 61 to 72% have a personality disorder [57, 58].

2.2.3.1 Severe MUS: Somatoform Disorders

DSM-IV classiﬁcation of MUS includes somatization disorder, undifferentiated
somatoform disorder, conversion disorder, pain disorder, hypochondriasis, and body
dysmorphic disorder [5]. The most validated of these entities, somatization disorder,
comprises less than 1% of all MUS patients in primary care [6, 39, 42]. In an attempt to
increase detection of severe MUS, investigators have deﬁned and studied 2 derivative
entities, abridged somatization disorder [[43, 59-61] and multisomatoform disorder [62,

63].

2.2.3.2 Severe MUS: Functional Somatic Syndromes

This includes medical syndromes that are characterized more by symptoms,
suffering, and disability than by disease-speciﬁc, demonstrable abnormalities [32, 46,
64]. Examples of these syndromes are multiple chemical sensitivities, sick building
syndrome, and the Gulf War syndrome. More established. syndromes are ﬁbromyalgia

[65], chronic fatigue syndrome [66] and IBS [67].

3. Frequency and Cost of MUS

The prevalence of all MUS in the outpatient setting ranges from 25 -— 75%
depending on the deﬁnition used [68, 69]. One study of ambulatory patients could
identify organic disease in only 16% of ambulatory patients with new symptoms, even
though diagnostic tests were performed in 70% of them [2]. A follow—up prospective
study revealed that the majority of patients (~70%) improve after 2 weeks and that this
improvement was sustained after 3 months [70], suggesting that the majority (~80%) of
these MUS patients had mild MUS [47]. One study found that 51% of primary care
patients with an average of 5 or more visits per year had moderate MUS and 14% had
severe MUS [41]. The prevalence of severe MUS ranges from 4.4 —— 22% in the
community to 33% in the outpatient setting [41].

The cost of healthcare for MUS is estimated at more than $256 billion /year [68].
This does not account for excessive utilization in mild MUS or the costs of loss wages

and early retirement.

. Etiology of Medically Unexplained Symptoms

The notion that one can have genuine physical symptoms that are unexplainable
[71] is problematic for both clinicians and patients. Patients require speciﬁc mechanisms
to help them understand their illnesses, to achieve legitimate sick roles, to access support
and resources, and to take actions on their illnesses. Similarly, clinicians need to explain
and treat distress, make appropriate referrals, and to recognize and process the threat of
MUS to their sense of competence. Understanding the following proposed mechanisms of

MUS has therefore been helpful to many clinicians.

4.1 Psychological explanations

In his comprehensive review, Brown [72] identiﬁed three groups of psychological
explanations of the pathogenesis of MUS based on dissociation, conversion and
somatization. Dissociation is a mental process that produces a lack of connection in a
person’s thoughts, memories, feelings and actions by reducing attention to certain
sensory channels, usually in response to traumatic experiences. Supported by cognitive,
electrophysiological and neuroimaging studies, modern dissociation theories assert that
MUS arise when normally adaptive dissociative process becomes overgeneralized [72].
Furthermore, children who were exposed to models of illness behavior, like an ill parent,
or those who were exposed to physical or sexual trauma are more susceptible MUS than

those who did not have similar exposures [73].

4.1.1 Conversion theories [72]

Conversion theories extend dissociation model by describing the way in which
emotional and motivational factors contribute to MUS generation. Unconscious
suppression or repression of traumatic memory purportedly protects the individual from
potentially overwhelming negative affect. To preserve energy balance, the brain
“converts” the repressed, negative affect into a somatic symptom that either was present
at the time of the initial trauma or is some symbolic representation of it. Thus the primary
motivation for MUS is to reduce anxiety by allowing individuals to express underlying
psychological conﬂicts without consciously acknowledging them. Secondary motivation

arises from all other advantages associated with being ill, such as sympathy, attention and

avoidance of work. Conversion theories are supported by studies that associate
alexithymia [74] and reinforcement (secondary gain) [75] with MUS, although such

associations are not consistent across all studies [76, 77].

4.1.2 Somatization models [72]

These focus more on the process and less on the speciﬁc mechanisms of MUS as
demonstrated by Lipowski’s [78] deﬁnition of somatization, “a tendency to experience
and communicate psychological distress in the form of somatic symptoms and to seek
medical help for them.” Best articulated by Kirmayer and Taillefer [79], somatization
models postulate that MUS develop when emotional arousal or normal bodily sensations
produced by everyday physiological processes are misinterpreted as indicators of disease
leading to illness worry, catastrophizing and demoralization and adoption of the “sick
role.” This exposes MUS patients to social forces that reinforce their illness behaviors.
The somatization process is moderated by several factors including patients’ personality
structures, previous illness experiences, response of signiﬁcant others and healthcare

providers, work conditions, disability, insurance and compensation systems.

4.1.3 Integrative model

Brown proposes an integrative approach [72] in which symptoms are purportedly
caused by stored information in the cognitive system that disrupts the interaction between
conscious and preconscious aspects of information processing. The development of the
stored information often is driven by defensive reactions that mitigate traumatic effects.

Central to the creation and maintenance of MUS is excessive focus on symptoms,

10

ampliﬁed by abnormal illness belief and behavior, worry, negative affect, secondary gain
and abnormal personality features.

While psychological theories, especially integrative models like that of Brown
[72], may provide a useful scheme for organizing existing and future research, cultural
biases against the notion of embodied emotions and mental illness limit the acceptability
of psychological explanations to patients [31, 80, 81]. Qualitative studies suggest that
MUS patients instead prefer tangible (physical) explanations that they can understand and
that free them from blame [82]. Consistent with this, Sharpe and others [31] have
proposed a paradigm shift in which MUS is “remedicalized” around psychophysiologic
explanations. These theories simultaneously acknowledge psychological and
neurobiological correlates of physical symptoms by highlighting reversible functional

disturbance, rather than ﬁxed psychological pathology.

4.2 Psychophysiologic explanations

Psychophysiologic theories provide plausible explanations for most common
physical symptoms [3 l , 71]. Bi-directional ﬁbers make emotional and cognitive input
from the brain possible in the perception of symptoms. Disturbed regulation of
physiological systems caused or aggravated by psychosocial stress or limitation causes
MUS. These perturbations do not require structural lesions detectible by current clinical
examinations or diagnostic tests. A classic example is provided by the Gate Control
Theory of pain, the basic premise of which is active CNS modulation of pain from
peripheral nerves through a network of bidirectional pathways. This ‘gating system’ can

be affected by many psychological factors known to affect the brain, like current emotion

ll

and prior experiences with pain and anxiety [83, 84]. Pain disorders purportedly result
from disruption of normal CNS response to sensory stimulation that alters patients’
perceptions and interpretations of normal bodily sensations [85]. Functional and
structural brain imaging as well as other preliminary physiological studies support
mechanisms in other MUS syndromes like chronic fatigue syndrome and ﬁbromyalgia
[86], and IBS[67].

In all the mechanisms discussed thus far, the etiology of MUS is thought to
originate in the individual. This may be problematic in some patients such as those who
belong to groups where social values are elevated above individual autonomy. For these
patients, sociosomatic theories which are supported by ethnocultural studies provide an

alternative framework for explaining MUS [71].

4.3 Sociosomatic explanations

Sociosomatic explanations locate the etiology of symptoms in societies rather
than individuals [71, 87, 88]. Many social groups have developed culturally acceptable
language that embody combinations of physical, emotional and social meanings and offer
clues to accepted meanings of certain symptoms [87]. For example well-described
culture-bound syndromes like “ataque de nervios” among Hispanics and “hwa-byung” in
Koreans are understood as resulting from cumulative anger repressed to maintain social
harmony and relationships [89-92]. Other symptoms serve as a form of social protest or
challenge that helps to mobilize appropriate resources while protecting individuals from
the consequences of complaining about one’s psychological or social plight. For

example, illness narratives of Vietnamese immigrants attributed the etiology of their

'

12

MUS “uat u’c” to indignation over a social injustice that had to be endured in order to
maintain social order and harmony [71]. The study provided the ﬁrst opportunity for
many of these participants to discuss their interpretations [71]. Mindful clinicians can
offer patients with MUS similar opportunities to create meanings for their symptoms by

exploring their characteristics,

5. Risk factors for MUS

Several studies have shown a high association of MUS with anxiety and
depression. In a large multinational cross-sectional survey of primary care patients,
Gureje et al. [93] showed that patients with somatization disorder were more likely to
have depression in 13 out of 14 countries (OR 2.54 — 7.07, p<0.001). Similarly, anxiety
was more prevalent among patients with SD in 13 out of 14 countries (OR 2.20 — 7.34).
These ﬁndings are consistent with data from the Epidemilogical Catchment Area trial [4],
which showed that most symptoms in community patients were associated with at least a
twofold increased lifetime risk of depression and anxiety. Katon et al. [55] enhanced the
documentation of this association by demonstrating a “dose-response” relationship. In an
attempt to test their hypothesis that MUS represents a spectrum rather than a dichotomy,
they divided distressed high utilizers who were randomized to receive psychiatric
intervention into four categories according to the number of medically unexplained
somatic symptoms. Their results, which are summarized in Table 1, show that the burden
of psychiatric illness and disability increases with increasing number of unexplained

symptoms. A major limitation of all these studies is their cross-sectional design, which

13

precludes conclusions about the directionality of the association between MUS and
anxiety or depression. Does one cause the other, or is one a subset of the other?

If one causes the other, which is the

 

 

 

 

 

 

Table 1: Prevalence of Lifetime Anxiety and
. _ 9 Depression in High Utilizing Patients with MUS
cause and Wthh rs the effect. None Group Panic Major Dysthymic
. . . . (Number of Disorder Depression Disorder
of the studies reportrng assocratrons symptoms; n) N CV) N (%) N (%)
0
_ 1 (women <
can answer these questlonS- 6, men <4; 1 (3.2) 14 (45.2) 7 (22.6)
31)
Inconsistent results of trials of 2 (women: 6-
8, men 4-8; 8 (20.0) 30 (75) 9 (22.5)
. 40)
treatment of MUS syndromes With 3 (9 _ 12; 21)
. . _ 4 (19.0) 15 (71.4) 5 (23.8)
antidepressant medication also 4 (> 13;
(N=27) 13 (48.1) 22 (81.5) 17 (63.0)
highlight the lack of clarity about the

 

 

 

 

 

 

nature of the relationship [94, 95].

In addition to depression and anxiety, childhood and adult violence are emerging
as important risk factors for MUS [96-102]. A cross-sectional survey [100] of 1931
women who sought care in 4 community-based internal medicine clinics revealed more
physical symptoms (mean :SD 6.2 i 0.2 vs 4 i 0.9, p<0.001) and higher mean MUS
score (p<0.001) in patients with previous childhood sexual or physical abuse. The
prevalence ratio of previous childhood abuse was 3.5 (CI = 2.3 — 5.1) in women with the
highest scores (upper third) for MUS compared with those the lowest (lower third)
scores. Although this study had several limitations such as possible selection as well as
recall bias, it has important strengths that help to validate its results. Firstly, the study was
designed speciﬁcally to assess the relationship between physical / sexual abuse and
subsequent MUS and psychological illness. Their a priori hypothesis formulation

provided some protection against chance ﬁndings. Secondly, study participants were

14

demographically and socio-economically diverse, thus increasing generalizability.
Thirdly, the strength of the association was relatively large, thus diminishing the
likelihood of important confounders. Finally the association is consistent with other
studies [96, 97, 99, 101], whose results are summarized in Table 2. Lessons learned from

these studies may prove useful in the design of MUS prevention and treatment studies.

 

 

 

 

 

 

Table 2: Summary of Studies on Sexual and Physical Abuse
Study Study setting and design Main ﬁndings
(reference)
I [101] Case-Control: somatization Higher childhood molestation
disorder compared with in SD
primary affective disorders
11 [96] Case series; 7 patients with Mean of 18 medical visits, 8
history of sexual abuse operations, 66-70% normal
ﬁndings with visits and
operations
III [97] Cross-sectional: functional vs. Functional disorders associated
organic disorders, consecutive with: sexual abuse (OR 2.08.
women in specialty clinic. CI = 1.03 — 4.21) and physical
abuse (OR 11.39, CI = 2.22 —
58.48)

 

 

 

 

6. Diagnosis MUS in primary care

Effective treatment for MUS in primary care exists [103], but often is not utilized
as both healthcare providers and patients are caught in a cycle of fruitless diagnostic
testing and unsatisfactory symptomatic treatments. Diagnosis of MUS is essential to
limit excessive diagnostic testing and to initiate treatment of MUS.
6.1 Clinical Diagnosis
6.1.1 Mild MUS

Mild MUS is diagnosed primarily by obtaining an appropriately detailed history
and physical exam and observing the patient over time for resolution [47]. Most

symptoms in primary care resolve spontaneously without being medically explained. For

15

example, review of the charts of 1000 ambulatory care patients revealed an organic
cause in only 16% of patients even though diagnostic tests were performed in 70% of
them [2]. Similarly, a prospective study of 500 ambulatory clinic patients showed that
70% of patients improved after two weeks and that this improvement was sustained after
3 months [70].Ordering unnecessary tests that come back negative can heighten patients’
concerns that a serious disease is being missed. False positive tests can lead to further,
sometimes harmful testing and can increase anxiety. Therefore, for low-utilizing patients
with acute or new symptoms in whom organic disease is unlikely following an
appropriately detailed history and physical exam, a diagnosis of mild MUS can be made.
The psychosocial context of the patient’s symptoms must be determined to ascertain
stress or to help diagnose those patients whose symptoms may be due to their
psychosocial circumstances. Rather than tests and consultations, clinicians rely upon

observation over time for prompt resolution.

6.1.2 .Moderate to severe MUS

In contrast to mild MUS, moderate to severe MUS must be evaluated by
appropriate testing and consultation to exclude organic disease. There is a high
prevalence of organic disease in patients with chronic, disabling symptoms, initially felt
to be due to MUS because there were no objective ﬁndings on history and physical
exams. For example, Laparoscopic studies in 100 women with pelvic pain for at least 6
months revealed organic pathology including endometriosis, adhesions, and others, in
83% [104]. Similarly, ten (11.8%) of 85 patients referred to a psychiatric hospital with a

diagnosis of conversion disorder were found to have a neurological disorder after median

16

follow-up period of 2.4 years [105]. False positive rates for conversion disorder have
been found to be as high as 15% in a comparable study with 10 years of follow-up [106]
and as low as 4.7 % in a 6-year follow-up study conducted at secondary and tertiary
neurological centers [107]. Clinicians must maintain a high level of suspicion for
conditions like multiple sclerosis, Lyme disease, systemic lupus erythematosus,
porphyria, celiac sprue) can elude diagnosis if the physician has a low index of suspicion.
In addition, many functional somatic syndromes (e.g., IBS) cannot be distinguished from
organic disease (e. g., inﬂammatory bowel disease), based on symptom criteria alone. For
instance, in a study of 602 new referrals to a gastroenterology clinic, the Rome criteria
yielded a sensitivity of 85% and a speciﬁcity of 71% for diagnosing IBS [108]. In another
study that used both retrospective and prospective designs, adding the absence of red
ﬂags to the Rome criteria yielded a sensitivity of 63% and a speciﬁcity of 100% in the
retrospective study, and a positive predictive value of 98% in the prospective study.
However, the study used gastroenterologists’ evaluation as the ‘gold standard’ and only
45% of patients 45 years or younger had their colon investigated [109].

Complicating matters further, MUS can occur with co-morbid physical and/or
psychological disease. Patients with co-morbid MUS are more severely ill than expected
from their physical disease. A medically unexplained symptom often may co-exist with
an organic disease (e. g., IBS with Crohn’s disease) that will respond to different
treatments; therefore, it is important to diagnose both co-morbid conditions.

However, one must guard against making a diagnosis of moderate to severe MUS
because prominent psychological features are present. In a prospective study of 97

referred patients that sought to determine the utility of six psychosocial factors for

17

diagnosing IBS, Smith et al.. [1 10] found that the psychosocial factors did not distinguish
between IBS and organic disease. Instead, psychosocial factors were related to high
utilization in both IBS and organic disease. The authors concluded that psychosocial
criteria were of limited value in differentiating IBS from organic disease.

Once deﬁnitive work-up has been completed, however, it is not necessary to
repeat them. Most excessive diagnostic testing is instigated by the doctor rather than, as
commonly believed, by the patient. This appears to satisfy the doctor’s diagnostic need
“to be sure” but, at the same time, it inadvertently avoids the patients’ expressed and

hidden psychosocial and emotional needs [111].

6.2 Identiﬁcation of M US patients for research

Our group has developed and published a reliable chart rating procedure to
identify high utilizing patients with MUS for research [44]. We clariﬁed symptom
categories from the charts of ambulatory patients with eight or more visits per year and
developed decision rules so that all symptoms could fall into a category and all categories
were mutually exclusive. We trained three senior internal medicine residents and two
primary care faculty members to rate charts to determine whether an organic disease was
identiﬁed for all recorded symptoms in the year before a randomized controlled trial of
treatment for patients with MUS. The raters classiﬁed each physical symptom as
‘documented organic disease’ if deﬁnitive tests such as laboratory, radiographic or other
diagnostic testing, classic or deﬁnitive physical ﬁndings and/or consultants’ opinions
based on objective testing provided an organic disease explanation; if tests were negative,

symptoms were rated “documented non-organic disease”. Symptoms that had either

18

insufﬁcient or no testing were rated ‘undocumented disease.” Raters then summarized
each visit based on the number of categories represented and added all categories in the
12 month-period. Percent MUS/visit was deﬁned as the sum of documented organic and
undocumented categories divided by the total number of categories over 12 months.
Inter-rater reliability was high — percent agreement for documented organic, documented
non-organic and undocumented were 92%, 96% and 92% respectively. We used this
method to identify patients with MUS, and to quantify the proportion of MUS/visit over
12 months and their medical co-morbidities.

Because of difﬁculties previously mentioned, an alternative method of MUS case
identiﬁcation, like this validated chart review method, may be more effective than the
DSM-lV-oriented structured interview [44] or widely used screening instruments like the
Symptom Checklist 90, Revised (SCL-90-R) in patients with multiple outpatient visits.
The SCL-90-R is limited in the diagnosis of MUS in patients with chronic medical
diseases because it contains many items that reﬂect symptoms possibly attributable to the
medical disease [1 12]. The method is able to determine whether an organic disease was
identiﬁed for any recorded symptom and to classify MUS as documented non-organic
(appropriate tests, no organic disease) or undocumented (no documented test) MUS.
Because it does not rely on patient recall, it may be more accurate than a structured
interview like the diagnostic interview schedule (DIS) that requires patient recall of

lifetime prevalence of symptoms[44, 1 13].

19

7. Treatment of Medically Unexplained Symptoms in Primary Care

Patients with moderate to severe MUS are likely to be very concerned, angry, or
sad about their symptoms and are potentially mistrustful of the clinician because of
previous experiences or because of worry about serious organic disease. Thus, empathic
interactions with clinicians enhance the patient’s participation in the treatment plan and
improve outcomes. A systematic review of 23 analytical and/or randomized controlled
studies found that patient-centered communication interventions reduced patients’
symptoms, increased satisfaction, and improved functional status and adherence to
medical recommendations [114]. However, most of the studies included in the review
involved patients with organic disease rather than MUS. In a RCT [115], resident
physicians trained in a 5-step, 25-substep patient-centered method that used an empathic
method called NURS (naming, understanding, respecting and supporting emotions), were
more likely to report increased conﬁdence in their psychological sensitivity and
management of MUS than resident controls. The study also showed a trend toward
improved patient satisfaction and health in the patients of trained residents compared with
those of resident controls. A review of the literature indicated that most trials of
interventions for MUS rarely attended to the provider—patient relationship systematically
[116].

Recently, primary care providers used the 5-step, 25-substep method to motivate
patients with moderate to severe MUS to engage in individualized treatment plans. This
RCT [103] demonstrated statistically and clinically signiﬁcant improvements in patients’
satisfaction with the providers’ communication and relationship building skills.

Satisfaction was in turn associated with adherence to the treatment plan (i.e., more

20

intervention patients decreased use of controlled substances (p=0.043) and more
increased use of full dose antidepressants (p = 0.037) compared with controls).

In addition to an effective relationship, the treatment employed proven cognitive
behavioral techniques. A systematic review [117] of 31 controlled trials (29 randomized,
2 nonrandomized), revealed that specialty conducted CBT signiﬁcantly improved
physical symptoms in 20 (71%) of the 28 studies that assessed them, and showed a trend
toward improvement in another 3 (11%). Similarly, CBT deﬁnitely (n=9, 47%) or
possibly (n=5, 26%) improved functional status in 17 studies. In contrast only 38% and
8% of 26 studies showed deﬁnite or possible improvement respectively in psychological
distress with CBT. Beneﬁts obtained at the end of treatment persisted at follow-up
(range: 1 month — 24 months) in 29 of 30 studies that studied durability of outcome.
Smith et al.. [103] demonstrated that primary care providers could use similar cognitive
behavioral techniques of education, commitment, goal-setting and negotiation to treat
MUS. In their study [103], 48(49%) intervention patients compared with 34(33%) usual
care patients achieved the primary endpoint of a 4-point improvement in the mental
component summary (MCS) of the SF-36 (OR =1.92, CI = 1.08 — 3.40). According to the
authors, a 4-point improvement in MCS corresponded with the improvement observed
following combined mitral and aortic valve replacement.

An important part of MUS treatment is to motivate patients to adopt healthy
lifestyle behaviors. Healthy behaviors enhance the effectiveness of other aspects of the
patient’s speciﬁc treatment plan, and improve the MUS patient’s function and quality of
life. Many of the recommended activities have some, though not optimum, supporting

evidence. For example, a non-randomized controlled trial [1 18] of a convenience sample

21

of 16 patients showed that 15 minutes of relaxation response meditation twice a day for
six weeks reduced symptoms recorded in symptom diary, especially pain and bloating, in
patients with IBS. The effects of the program persisted after 1 year of follow-up [119].
Similarly, a prospective, randomized trial [120]of 58 patients revealed that swimming
pool exercises matched to patient’s threshold of fatigue and pain signiﬁcantly improved
Fibromyalgia Impact Questionnaire score and 6-minute walk test compared with usual
care controls. Follow-up was after 6 months. In addition to better improvement in the
primary end-point, the RCT trial by Smith et a1..[103] demonstrated signiﬁcant
improvement in disability scores of intervention patients (p = 0.001 ), but not in the scores
of control patients (p = 0.26).

In addition to this non-drug therapy, research also supports some pharmacologic
agents, but not others. Narcotics and other addicting medications may cause depression
and exacerbate the patient’s symptom, while non-narcotic analgesics and other benign
symptomatic medications are often effective and inexpensive. Antidepressants are useful
for both symptom reduction and treatment of co-morbid depression and anxiety. In a
systematic review [121], 64 out of 94 trials (69%) demonstrated some beneﬁt of
antidepressants in patients with MUS. Patients receiving antidepressants were more than
3 times as likely to experience symptomatic improvement than patients receiving
placebo. Meta-regression did not differentiate effect across different classes of
antidepressants, however studies of tricyclic antidepressants (TCAs) were more likely
than studies of selective serotonin re-uptake inhibitors (SSRIs) or antiserotonin agents to
have a beneﬁcial outcome (p=0.02). Depression was assessed in only 49 (52%) of studies

and association between depression and response to treatment was performed in only 24

22

studies (25% of all studies). Of the 24 studies, only 8 (33%) demonstrated a correlation
between physical symptom response and depressive response. Moreover, the study was
plagued with signiﬁcant publication bias. In Smith et al.’s study [103], 65 (68.4%)
intervention patients, compared with 20 (19.8%) usual care controls increased use of
antidepressants to full doses (p<0.001). Similarly among patients who were previously
using controlled substances, 26 (70.3% of using intervention patients) compared with 6
(14.3% of using control patients) reduced usage (p < 0.001). Increase in use of full anti-
depressant (p = 0.001), but not reduction in use of controlled substances (p=0.26), was
associated with improvement in the primary endpoint of the trial [103]. Thirty-seven
(80%) of patients who improved took full dose antidepressants, suggesting that full dose
antidepressant use was a signiﬁcant, but not the only contributor to improved patient
outcome. There is consensus that TCAs may have more analgesic efﬁcacy in chronic pain
than SSRIs mainly because more studies involve TCAs and studies involving TCAs more
consistently demonstrate efﬁcacy against pain [122].

Physicians commonly experience negative emotional reactions to patients with
MUS. These negative emotions can be harmful to patients and lead to physician ‘burnout’
if not recognized and addressed. Moreover, physicians’ personal characteristics, past
experiences, and biases can have important effects on communication with patients,
especially those with MUS. A qualitative study [123] revealed that maladaptive behaviors
like being overly controlling, avoiding psychological material and being passive or
detached were common during medical residents’ and fellows’ interactions with patients.
These behaviors were usually associated with previously unrecognized emotions like

anxiety about losing control, anxiety about addressing psychological material, or anxiety

23

about harming the patient. lnfrequently, discussions with these trainees uncovered
feelings of sexual attraction, anger, disdain, feelings of inadequacy or intimidation that
may have been rooted in personal issues and previous experiences. Another qualitative
study showed that 6/53 resident learners had personality patterns that inhibited self-
awareness work [124]. These learners showed little learning and little clinical use of the
patient-centered methods being taught. It may be occasionally necessary for clinicians
with similar personality patterns and attitudes to transfer care of MUS patients to other

colleagues.

8. Summary

MUS is a difﬁcult and common problem in primary care. We have made
signiﬁcant progress over the last ten years. We have recently published a proposed
classiﬁcation scheme that may be more useful to clinicians. It is awaiting empirical
testing. We have also developed an effective treatment that can be deployed by primary

care clinicians. The ﬁeld needs descriptive data on MUS in primary care.

24

CHAPTER TWO

Structural and Functional Neuroradiological Changes in Patients with Medically

Unexplained Symptoms (MUS) Syndromes: A Systematic Review

Francesca C. Dwamena, MD

25

1. Introduction

Medically unexplained Symptoms (MUS) refer to physical symptoms that
suggest, but are not fully explained by, a general medical condition; or the direct effects
of a substance or by another mental disorder. [5] In the psychiatric literature, these
“functional somatic symptoms” [125]and “somatization symptoms” [126] are
incorporated into the nomenclature as “somatoform disorders.” Some medical specialties
have also developed “functional syndromes” such as IBS, chronic fatigue syndrome, and
ﬁbromyalgia. These conditions share common features including symptoms like fatigue,
pain, disability out of proportion with physical examination ﬁndings and an association
with stress and psychosocial factors. Indeed, the overlap between these syndromes [32,
127] has led some authors to suggest that they are different manifestations of the same
basic problem, [34, 35, 37, 64] whose cause and pathophysiology remains enigmatic.
Because many of these patients complain of headaches and show some cognitive
dysfunction, the brain is believed to be a good candidate for abnormality.

In addition to laboratory studies, investigators have pursued brain defects in some
of these syndromes by conducting structural and functional neuroradiological studies.
Although both computer assisted tomography (CT), and magnetic resonance imaging
(MRI) permit reliable in vivo structural imaging of the brain, MRI appears to be more
sensitive in detecting abnormalities in at least one MUS syndrome [128] and is chosen for
evaluation in this paper. More recently, more sensitive functional imaging techniques
including single-photon emission computed tomography (SPECT), positron emission
tomography (PET), and functional MRI have been developed and are being employed in

the study of MUS syndromes [129, 130]

26

The purpose of this paper is to summarize studies of structural and functional
neuroimaging using MRI, functional MRI, PET and SPECT in patients with MUS. We
also sought to examine whether there were common patterns of neuroradiological

ﬁndings among studies of MUS syndromes, and suggest directions for future research.

2. Methods
2.] Literature retrieval

English-language articles were identiﬁed through a search of the SilverPlatter
Medline database from 1966 to June 2002. Search terms and strategy included (magnetic-
resonant-imaging/all subheadings, PET scan or positron emission and tomography-
emission-computed/all subheadings) and (“Somatoform-Disorders "/all subheadings or
“hypochondriasis "/all subheadings or “hysteria ”/all subheadings or “fibromyalgia "/all
subheadings or "Fatigue-Syndrome-Chronic ”/all subheadings or “Food-
Htpersensitivitv "/all subheadings or “Colonic-Diseases—Functional "/all subheadings).
Variants of these terms were also included by using the ‘explode ' function. Additional
keywords included functional somatic symptom(s) andfunctional somatic syndrome(s).
Finally, the bibliographies of retrieved reports were examined for other articles. This
process yielded 27 articles, 11 of which are included here. A meta-analysis could not be
conducted given the small number of studies and because of variations in study design,

methods, and case deﬁnitions.

27

2.2 Inclusion Criteria

Original articles describing neuroradiological studies in patients with MUS are
included in this article. Only 1) full articles published in peer review journals in the
English language are included. To ensure adequate comparisons, we required that 2)
radiological studies should be done in all study subjects. Other inclusion criteria are 3)
adequate number of patients (_>_ 10), 4) at least one control group and 5) a clearly deﬁned
patient population. One original study [131], which assessed MRI abnormalities in
patients with chronic fatigue syndrome did not have radiological studies in all patients
and was excluded. Another study [132], whose main goal was to assess cognitive
functioning and MRI ﬁndings in chronic fatigue was also excluded because patients did
not have to meet criteria for CFS, or any other MUS syndrome. Two studies [129, 133]
were excluded because they evaluated patients with MUS syndromes but had no controls.
All other studies were excluded either because they were not original studies or did not

address this study’s question.

2.3 Recorded Variables

Recorded variables from each study included the full study reference, type of
MUS syndrome, type of radiological study, number of patients, number of healthy
controls, type and number of other controls, central nervous system site and abnormalities
reported, and the major ﬁndings. The following variables were also extracted to assess
the quality of the different studies: Sampling, study design, gender distribution of
subjects, mean age, and whether or not the one reading the radiological study was

bhnded.

28

3. Results

The qualities of the studies selected for inclusion in this report are summarized in
the Table 3. Most of the studies were controlled cross-sectional studies of convenience
samples. About half of the studies documented blinding of the radiologist that reviewed
the neuroradiological study. There was however some variation in the quality of studies
as shown in the table.

Table 4 summarizes the major ﬁndings from the studies. Seven and four of the
eleven studies involved chronic fatigue syndrome (CFS) and IBS (IBS) respectively.
There were no suitable studies of ﬁbromyalgia, somatization disorder or other
somatoform disorders, hypochondriasis or any of the other MUS syndromes included in
our extensive literature search. Studies of CF S evaluated both structural as well as
functional abnormalities while all IBS studies were investigations of ﬁrnctional
abnormality. IBS studies all involved neural responses to rectal stimuli.

Although there was considerable variability among studies within each MUS
syndrome, all but one study documented significant functional abnormality compared
with normal and other controls. Because the methods employed in IBS studies were
similar among each other, but completely different from CFS studies no comparisons
could be made between the two groups. Thus, I have made only within group

comparisons.

3.1 Chronic Fatigue Syndrome (CSF) studies

Five of the seven CF S studies used functional neuroradiological studies (mostly
SPECT). Most of the abnormalities involved the frontal, temporal and parietal lobes, but
one relatively large study documented brainstem hypoperfusion in all CF S patients
compared with normal and depressed control patients.

There were 3 MRI studies of structural changes in CFS. All of these showed
increased subcortical white matter signal intensities compared with controls. Most of the
abnormalities, like the functional studies, involve the frontoparietal lobes. Other changes,
which were not consistent among the studies, include increased intensities in the corona

radiata, centrum ovale, frontal white matter and periventricular areas.

3.2 Irritable Bowel Syndrome (IBS) studies

Three of the four studies used functional MRI while two used PET scans. One
study used both. Again, there was signiﬁcant variability but all the studies showed
enhanced anterior cingulate gyrus activation with rectal distension compared with normal
controls. Other areas of abnormality that are not consistent among studies include the left
prefrontal cortex, left somatosensory cortex, the brainstem, temporal lobes, insular

cortices and the thalamus.

4. Discussion
The most important ﬁnding of this systematic review of the literature on structural
and functional neural abnormalities in MUS syndromes is that very few studies have been

conducted. All studies that met inclusion criteria involve either CFS or IBS. Similar

30

studies must be conducted in patients who meet criteria for ﬁbromyalgia, somatoform
disorders or other MUS syndrome if the possible common etiological factor of these
syndromes is to be fully elucidated.

The review also shows that although the current literature precludes comparison
of neuroradiologic changes between different MUS syndromes, there are some common
observations from studies within groups. Patients with CFS appear to have signiﬁcant
abnormalities in the subcortical white matter of the frontal, parietal and temporal lobes
when compared with controls. This is not surprising given the fact that there is
considerable cognitive dysfunction in patients with CF S. It will be interesting to see if
similar changes are documented in other MUS syndromes. It must be emphasized,
however, that there was considerable variation in other areas of abnormality among the
CFS studies.

Studies of IBS also showed remarkable consistency in abnormalities involving the
anterior cingulate gyrus. The reported alterations may be related to altered central
noradrenergic modulation and are consistent with ﬁndings of emotional lability in
patients with IBS. It is quite possible that these ﬁndings are common to other MUS
syndromes or psychological disorders but this has not been studied.

The ﬁndings of this review must be interpreted with consideration of its
limitations. Although considerable effort was made to ﬁnd all relevant articles, it is quite
possible that important studies were missed. In order to improve comparability, I did not
include studies with CT scans, but this may have systematically excluded important
information about structural abnormalities in these patients. Furthermore, broadening the

search to include studies with CT scans may yield information on other MUS syndromes

31

such as ﬁbromyalgia and somatoform disorders, thereby permitting comparisons between
the groups.

These limitations notwithstanding, this preliminary review is the ﬁrst attempt to
summarize and compare information on structural and ﬁrnctional abnormalities in
different MUS syndromes. It provides a framework for a more complete literature search
and subsequent review, whose data can inform the search for the pathophysiology of the

vexing problem of MUS.

32

 

Table 3: Systematic Review - Study Demographics and Quality

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

Study (Ref) Sampling Case Deﬁnition Study Gender Mean age Blinded
Desiﬂ review?

Schwartz et Convenience CDC, British or Cross- 64% female 70 i 13.2 Not stated
a1.[128] sample, Australian sectional

healthy deﬁnition of

volunteers chronic fatigue

syndrome (CFS)

Ichise et al.. Convenience Clinically Cross- Yes
[1 34L sample deﬁned sectional
Schwartz et Convenience CDC, British or Cross- 69% female 42.5 i 2.3 Yes
al.. [135] sample, Australian sectional (SPECT)

healthy deﬁnition of

volunteers chronic fatigue 62% female 49.6 i 6.2

syndrome (CFS) QVIRI)

Costa et al. Convenience, Clinical Cross- 67 40 Not stated
[136] healthy diagnosis sectional

volunteers
Lange et al. Convenience 1994 case Cross- 80% female 36 i 8 Yes
[1 3 7] sample deﬁnition sectional
Tirelli et al. Convenience CDC deﬁnition Cross- 65% female 34 j; 15 Not stated
[138] sample for CF S sectional 48 i- 7 (dep)
Greco et al. Convenience Clinical Cross- 75% female Range 22 - Yes
[138] sample diagnosis sectional 78
Silverrnan et Cosecutive Rome criteria for Cross- Not stated Not stated Not stated
al. [139] sample from IBS sectional

referral center
Bernstein Convenience Clinical Case 67% F in 40.2 i 6.6 Not stated
[140] diagnosis series cases

100% males 33.8 19.4
in controls

Naliboff et Convenience Rome criteria Cross- 16% female 39 (27 — 58) Not stated
al.[l4l] sectional
Mertz et a1 Convenience Rome criteria Cross- 90% female 33 (20 - 52) Yes
[142] sample sectional

 

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36

CHAPTER THREE

In Their Own Words: Qualitative Study of Hi gh-Utilizing Primary Care Patients with

Medically Unexplained Symptoms

Francesca C. Dwamena, MD; Judith S. Lyles, PhD; Richard M. Frankel, PhD;

Robert C. Smith, MD, 80M.

37

ABSTRACT

Objective: To describe perceptions and experiences of primary care patients with
medically unexplained symptoms (MUS).

Methods: A random sample of 19 high utilizing primary care patients, for whom 69.6%
of visits were medically unexplained, were encouraged to talk spontaneously about
themselves and answer semi-structured questions. Verbatim transcripts of interviews
were analyzed using Grounded theory.

Results: All but one participant described at least one type of negative experience.
Regardless of their experiences, all except three participants recounted stories of
achievement, positive action, and/or altruism. The three participants who did not report
any positive behaviors projected very low levels of insight and a sense of entitlement.
They talked oﬁen and vaguely about their symptoms and expected to be excused from
societal roles. Regardless of their level of insight, achievement, action, and/or altruism,
ﬁve participants who were worried about missed diagnosis complained about their
healthcare and focused on their symptoms.

Conclusion: Different perceptions and behaviors separated patients with similar negative
experiences. Lack of insight and a sense of entitlement were associated with symptoms
focus. Worried participants complained and focused on symptoms.

Practice implications: Rather than negative experiences, MUS patients’ levels of insight,

entitlement, and worry may inﬂuence illness and functional behaviors.

38

1. Introduction

Patients with medically unexplained symptoms (MUS) are common in primary
care [1, 144]. Caring for them can be expensive [145], and frustrating[l45-147].
Descriptive studies of MUS have traditionally relied on deﬁnitions of MUS derived from
the Diagnostic and Statistical Manual (DSM). However, recent studies show that 50 —
75% of all high utilizing primary care patients with MUS do not meet full or abridged
DSM criteria [41, 42]. Thus, descriptive studies that use DSM may not well represent
MUS patients seen in primary care. Moreover, qualitative studies with more liberal
deﬁnitions have focused on referral patients [82, 148, 149]. We conducted the study
reported here to better understand the perceptions and experiences of high utilizing

primary care patients with MUS.

2. Methods

We used Grounded Theory methods [150-152] to test, elaborate, and reﬁne
emerging categories from verbatim transcripts of 19 interviews to provide the analysis for
this study. The Institutional Review Board at Michigan State University approved the

study, and all patients gave informed consent.

2.] Subjects and Setting

We have described elsewhere how we identiﬁed patients for a randomized
controlled trial (RCT) of treatment for primary care MUS patients in a large health
maintenance organization in Michigan in January 2000 [103, 116]. Brieﬂy, trained

physicians used a reliable chart rating method [44] to identify high utilizing patients for

39

whom MUS accounted for at least 50% of visits. The study sample was a heterogeneous
group of patients with frequent minor complaints and patients with chronic, persistent
symptoms. We recruited a random sample of 19 control patients after they completed the
RCT to participate in this qualitative study. Participants of this study were similar to
other control patients in mean age (47.95 yrs vs. 46.26, P: 0.47), gender (84% female vs.
74%, P=0.55), and mean number of visits/year (12.79 vs. 13.26, p= 0.72), but had a

higher percentage of MUS visits (69.6% vs. 60.4%, p= 0.04).

2.2 Interviewing and Analysis

The interviewer (FCD) was trained in qualitative interviewing and had no prior
relationship with any participant. She told participants that the goal of the study was to
understand the experiences and perceptions of patients with multiple clinic visits. Using a
semi-structured questionnaire, (Appendix 1) she began each interview with open-ended
inquiry that allowed participants to determine the content, pace, and sequencing of the
interview. She noted and followed up important clues and tested, in later interviews of
different patients, theories that emerged from earlier ones. For example, after the ﬁrst two
interviews, she tested the emerging theme of childhood distress and history of abuse in
the remaining interviews, and, after analyzing the ﬁrst ﬁve interviews, she asked follow-
up questions whenever patients spontaneously brought up the topic of religion. Similarly,
she tested the notion of gender inﬂuence on doctor-patient relationships in subsequent
interviews when the narrative of one of the participants suggested it might be important.
If after 30 — 45 minutes the following a priori topics had not arisen, she asked about

them: participants’ explanatory models [153], locus of control [154], health-seeking

4o

behavior [155], relationships, and expectations for the future. All interviews were
audiotaped and were transcribed verbatim. The transcripts, stripped of patient identiﬁers,
were used to conduct further analysis.

We used an inductive process [150-152] to ensure that further analysis was
grounded in the data rather than based on our own pre-existing groupings or framework.
Three of the authors (FCD, J SL, and RCS) derived preliminary themes (see Table 5) by
independently reading, taking notes, and verifying concepts from the ﬁrst ﬁve transcripts.
We reconciled differences, clariﬁed, and reﬁned categories by consensus and then
developed working themes (see Table 5) by testing preliminary themes against a second
set of ﬁve transcripts. With the working themes in hand we read and discussed the
remaining nine transcripts to further identify, reﬁne, and elaborate previous themes and to
identify any new themes that emerged. Finally, we reread all 19 transcripts, developing
and testing relationships in categories, and independently veriﬁed our ﬁnal themes. We
completed our analysis when we had categorized all relevant text in the 19 transcripts and

could make no ﬁrrther modiﬁcations.

3. Results

We identiﬁed eleven ﬁnal themes. In Table 6, we have deﬁned the themes and
listed participants who projected them. Appendix A shows the degree (low, moderate or
high) to which each participant communicated each theme. Themes fell into three broad
categories: a) “Experiences” were participants’ actual descriptions of events that
occurred in their lives; b) “Perceptions” captured participants’ attitudes and/or insights; c)

“Behaviors” were actions of participants that were observed during the interview, or were

41

inferred from their narratives. Certain perceptions appeared to be connected with certain
behaviors. In general, negative perceptions coexisted with negative behaviors and vice
versa. We did not discern similarly discriminating relationships between experiences and

perceptions or behaviors.

3. 1 Experiences
This category consisted of three uniformly negative themes. All except Participant
2 had endured at least one of these negative experiences. Six participants had suffered at

least two of the negative experiences and four had experienced all three.

3.1.1 Negative experiences - Childhood trauma

Ten participants reported personal abuse, loss, and/or death of a loved one during
their childhood. An important feature of this theme was the emotional expression that
accompanied participants’ stories. For example, Participant 7 who was sexually abused
by her aunt’s husband at 8yrs old said simply, “I was so scared let me tell you. ”
Similarly, Participant 15, sobbed as she recalled the shame and isolation she felt about
her childhood abuse, “ When I was 7, I was molested by my dad 's bestfriend. They would
never have believed that; and I never talked about it until I was 4] for years I thought
it was my fault. ” Some stories were more notable for the absence of emotional
expression. Participant 4’s explanation for her calm demeanor as she talked about being
inappropriately fondled by her grandfather was, “I think I shut my feelings of?” quite a

while ago. My dad died when I was .10. ” She went on to share about other consequences

42

of her father’s death, “I didn’t have him while I was growing up through my teenage

)

years, I did a lot of self-destructive behavior. ’

3.1.2 Negative experiences - Adulthood abuse

Six participants were abused physically, sexually, and/or verbally as adults.
Participant 6’s story was typical, “I got knocked around... I was in the hospital at least a
couple of times... he kept me dragged down. I was nothing. ” Some experiences were
associated with physical symptoms. For example, Participant 14 was raped and abused
physically by her boyﬁiend when she was 16 years old. She subsequently developed
chronic pelvic pain. Similarly, Participant 6 who suffered abuse at the hands of two
consecutive husbands suffered with medically unexplained chronic musculoskeletal pain
for over 12 years. Participants 6, 15, and 19 had divorced their abusive husbands by the
time of the interview. Participants 12 and 17 suffered primarily from emotional or verbal
abuse. The following statement by participant 12 showed the toll that even verbal abuse
could have on the victim’s self—esteem, “He was the very controlling type, very “I ’m

smart you ’re stupid. It took me a long time to get myself together. ”

3.1.3 Negative experience - Family patterns of distress and/0r dysfunction

This theme captured notable familial patterns of illness behavior, or behavioral
illness in 16 participants. For example, seven of Participant17’s immediate family
members had reversal of gastric bypass surgery, and Participant 3 regularly visited the
same chiropractor with her four sisters. Similarly, according to participant 5, somatization

was a common response to stress in his family, and Participant 19 was worried about

43

being a hypochondriac like her mother. Others described the cost of familial alcoholism
(Participants 11. 14, and 15) and mental health problems (Participants 1, 8, 13, and 18).
This theme also captured expressions of primarily negative feelings about families and
close relationships. Participants 1’s simple statement about perceived parental discord
and general household chaos summed up a lot of participants’ reactions — “They would
scream and throw things... That was scary. ” Participant 1 also felt pressured by family
members’ unreasonable expectations - “It makes me feel as if I ’m not parenting to their

)1 H

expectations... My ﬁiends at work said, “You are driving yourself crazy. Others were
disappointed by parental favoritism (e.g., Participant 9 said, “they can never give me
anything materialistic that would equal what they sacrificed for [sister] ”), and/or neglect

(“they had an opportunity to take care of me... but they didn ’t and I have a lot of

strugglesfrom that. ”)

3. 2 Perceptions
There was one positive theme (insight) and two negative ones (entitlement and

worry) in this category.

3.2. .1 Positive perception - Insight

This theme characterized six participants’ demonstrated appreciation of the
relationship between symptoms and stress. Participants 5, 8, 11, and 15 stood out as being
particularly insightful. Participant 5 understood the psychological basis of his medically
unexplained symptoms — “ The symptoms I imagine, well, you know, is probably where I

started. ...I had spent a very strenuous weekend working in theyard, that type of thing, so

44

there were some physical aspects to what happened I think, but there were some
psychological aspects too, because my wife that very weekend informed me that I should
move out of the house. ” Participant 8, seemed to understand the importance of sharing
her feelings, “no one [in her family] ever talked... but I talked about everything, ” and
suggested a key to effective coping - “at least 5 0% has to be your attitude.” Participant
'11 shared how he had learned from his brother that “most of the problems you have
aren’t out in the world. They ’re up here (pointed to head), and if some day I can control
that, then I wouldn’t have the problems... the aches and pains come with that. ”
Participant 15 had been able to process growing up with an alcoholic father, childhood
sexual abuse, living with two abusive husbands, and many childrearing mistakes through
22 years of psychotherapy. The stories of these participants revealed the sources of their
insight as psychotherapy (Participant 15), wise counsel (Participant 11), and personal
reﬂection (Participants 5 and 8).

More commonly, however, participants appeared unconscious of the possible
effects of psychological stress. In particular, Participants 1, 4, and 6 were noted for
having very low insight. Participants 1 and 4 repeatedly coupled descriptions of their
symptoms with their jobs, housework, spouses, and/or children. They seemed completely
unaware of this and did not even consider the possibility of association between their
symptoms and aversion to any of their roles. Similarly, Participant 6 was abused, “he
became even more violent with me. He threw me on the ﬂoor and was choking me, "
but attributed her MUS to a concrete event from her past, “after a lady drove me off the
road, I hit the curb and I felt a real sharp pain, and appeared unable to imagine any

possible contribution from the concurrent abuse. When asked if she felt her husband’s

45

abuse might have contributed to her symptoms, she said, "I had no lingering pain from

anything he did... ”

3.2.2 Negative perception - Entitlement

Nine participants appeared to believe that their symptoms entitled them to special
considerations (see Table 6). They expected their families to assume responsibility for
household chores, and to be excused from their jobs. All participants labeled with very
low insight (Participants 1, 4, and 6) demonstrated this sense of entitlement. Conversely,
none with very high insight (Participants 5, 7, 8, 11, 12, and 15) conveyed entitlement.
Participants with entitlement offered their symptoms as excuses. For example Participant
6 gave the following reason for her absenteeism, “I ’m just gonna feel rotten today and
not do very much. I am in pain, the more I do, the worse the pain gets, but I will just take
the day oﬁ? ” These participants’ expectations led to a lot of angst in their families. For
example, according to Participant 16, “T here are days when you’re angry at either
friends or family because they still don ’t get it... [My husband] will say, ‘Well I don 't
understand why you couldn ’t carry those groceries My daughter has to help me with a
lot of that stuﬂf garbage and groceries and things... she ’s mentioned to one of myfriends
that she hated me because I was always in pain... she will say, “I ’m like your slave

because you are hurt. " "

3.2.3 Negative perception - Worry

Six participants were concerned about undiagnosed disease. With the exception of

Participant 3, none of these worried participants was characterized with entitlement. For

46

Participants 5, 7, and 19, the root of this concern appeared to be a strong family history of
cardiovascular disease. Similarly, Participants 3 and 12 were concerned about developing
cancer because of a strong family history of the same. None had explicitly discussed their
underlying concerns with their healthcare provider. Neither normal tests, nor doctors’
benign assessments of their symptoms reassured worried participants. Usually, either they
or someone they knew had been the victim of a medical error. For example, Participant

12 said, “I had severe chest pains last winter, and of course they did a bunch of testing
and they couldn ’tfind anything... I don’t know what caused it and that kind of bothers
me... it was scary; you only have one heart... I have this thing in the back of my head all
the time; they missed my mom’s cancer twice so medicine is not fallible. ”

For some of these participants, the current interview was the ﬁrst time they had talked
openly with anyone about their fear of unrecognized disease. Participant 7’s comment
hints at a possible reason for this — "No, No! They don’t spend time with you; they don’t.
They are always in a hurry. No participant felt to have very low insight conveyed an
impression of being worried. However, three highly insightful participants (Participants

5, 7, and 12) also were worried.

3.3 Behaviors

The remaining ﬁve themes fell under the behaviors category. Like perceptions,
this category included both positive (achievement, action, and altruism) and negative
themes (symptoms focus and complaining). There were notable relationships between

these behaviors and the perceptions described above.

47

3.3.1 Negative behaviors - Symptoms focus

The transcripts of 13 participants showed a pervasive emphasis on symptoms (see

Table 6). Narratives about symptoms were oﬂen long and vague. Participant 10’s
monologue demonstrated how ﬁ'ustrating this communication style in particular and
symptoms focus in general could be for providers:
“1 go in and I say, ‘I can ’t sleep, and it kind of comes and goes... ’ One thing goes out of
whack and I feel a lot of things so I come in a lot of times and I need to get them all in,
I need to tell her_everything that is going on... I think in the beginning I might have
overwhelmed her; but she now knows... that I do have a lot of things coming at me. ”

All participants labeled with entitlement were also categorized with symptoms
focus. For example, Participant 10 above blamed her symptoms for her constant tardiness
at work. Indeed, she fantasized about not working because a job “falls into something
where it just doesn 't work with me. "

Conversely, with the exception of Participant 5, none of the participants with
insight emphasized symptoms. The relatively few symptoms they talked about were more
concrete. Furthermore, their descriptions were concise. For example, Participant 15 said
of her back pain, “I would hurt within two hours [of bending over and underneath cars at
work]. ” Participant 5’s idiosyncratic symptoms focus may have been related to worry —
“I was really worried, that when you are in the situation you get, you do not know when
you are going to get better or not. You know, what is the rest of your life going to be like?
If] was so bad, at the relatively young age that I needed a cane. Would I be able to go

back to work? Would I have to go on disability? Would I be in pain all the time? "

48

Worrying was associated with symptoms focus in Participants 3, 5, 9, and 19. For
example, Participant 12 went to the doctor for “lots of ongoing little things ” because she
worried about cancer; and She said, “When something happens, you know, If I get a toe
injury or whatever of course the first thing you think is ”Oh my God! ” She insisted on
having ﬁve lumpectomies, even though her surgeon did not think they were indicated,
because “If they can make a mistake once, they can make a mistake again, and it isn ’t

gonna to be on me. I mean, I guess I feel like I have to be extra vigilant. ”

3.3.2 Negative behaviors - Complaining

This theme captured expressed and/or inferred dissatisfaction with healthcare
systems and/or providers. Four of the eight participants that displayed this theme
questioned the ﬁnancial motivation of their doctors and/or insurance providers. For
example, participant 9 wondered about one of her doctors, “Do you get an extra little
money in your pay check or something for ﬁnding diseases, I mean what "3 the scoop
here, you know? ” Similarly, Participant 7 thought the only reason she was expected to
see a nurse practitioner instead of a specialist for routine gynecological care was “to save
money, because that is the only thing, there is no other excuse, it is just to save money. "

With the exception of Participant 5, all worried participants complained about
some aspects of their healthcare. For example, according to Participant 7, “The nurse
sees you instead of the Gynecologist. She doesn’t order any ultrasound unless you ask...
Later she revealed that she preferred physicians and wanted tests because of concern
about a missed diagnosis. She believed tests were not being ordered because her

providers were ‘ffollowing some orientation [from insurance companies]. ” She continued

49

to share her emotional response, “I think this is bad... I feel helpless when it comes to
doctors because I don ’t know where to go to complain... Frustration is the biggest word
in my life, just about everything. ” With the exception of those who were also worried, no
insightful participant complained about the healthcare system. Similarly, none except two
entitled participants complained unless they were also worried, and no participant with

very low insight complained.

3.3.3 Positive behaviors - achievement, action and altruism

Sixteen participants shared about some signiﬁcant achievement of higher
education, leadership, professional status, entrepreneurship, and/or creativity (theme:
achievement). For example, Participant 8 wrote poetry and managed a local store while
she pursued a professional degree, and Participant 7 proudly operated an award-winning
website. Twelve of these participants also demonstrated ability to cope effectively and/or
make appropriate lifestyle changes (theme: action). For instance, Participant 12 retumed
to school after divorce and retrained for her current job. She was also careful to seek out
the best medical advice and follow it, and Participant 5 was able to quit smoking and
maintain a healthy diet. Nine participants demonstrated altruism in addition to
achievement and action. For example, Participant 8, who was a poet, actively pursued
pleasurable activities to help her cope with her illness, and she provided supportive care
for several family members - “I was there when [my stepfather] had his surgery... My
oldest brother suﬂ‘ers from post-traumatic stress syndrome... I have been doing a lot to
help him. ” Similarly, Participant 15 was able to quit a lifelong habit of drinking heavily,

quit smoking, and control her diabetes with diet. She also enjoyed her work and planned

50

to volunteer after her imminent retirement. Unlike participants who sought excuses to not
go to work, she actually felt ambivalent about retiring. Given the context of their negative
experiences, the positive attributes of some participants were remarkable. For example,
Participant 15 said, “I was raised in a very dysfunctional family; my dad had a bad
drinking problem when I was seven 1 was molested by my dad 's best friend But I
have now dealt with it [I quit drinking] because I was having black outs and it was
scaring me... I just quit. ”

Three participants (Participants 1, 4, and 6) did not demonstrate any of the three
positive behaviors. These same participants were rated very low on insight, and high on
entitlement. All three focused on symptoms, and instead of ﬁnding meaning in their
work, they seemed to focus on getting time off, or they quit their jobs. In contrast, all
participants who were worried, and or insightful demonstrated all three positive

behaviors.

4 Discussion And Conclusion
4. 1 Discussion

We achieved our primary objective of exploring the perceptions and experiences
of these high utilizing primary care patients with MUS. None of the themes uncovered
could be characterized as positive experiences. Instead, all but one, participants reported
at least one negative experience of childhood trauma, family patterns of distress, and/or
adulthood abuse. This is consistent with other studies [97, 156, 157] that have
demonstrated an association between the psychological distress of sexual / physical abuse

and MUS.

51

We also identiﬁed one positive (insight) and two negative (entitlement and worry)
perceptions that were associated with speciﬁc behaviors. Participants who demonstrated
psychological insight also shared stories of achievement, action, and altruism. Their
psychological grth resulted from psychotherapy, continuing wise counsel from a
relative, and or personal reﬂection. Unless they were also worried, these insightful
participants did not complain about their healthcare. In contrast, those felt to have very
low insight had no stories of achievement, action, or altruism. Instead, they perseverated
on their symptoms and projected a sense of entitlement. These participants resembled the
stereotypical somatization patients described by Pilowski and others [55, 78]. Consistent
with observations of alexithymia in patients with somatization disorder [158], they did
not complain or seem worried about undiagnosed organic disease.

The third perception identiﬁed was worry, usually resulting from a signiﬁcant
family history, previous knowledge of negative healthcare outcome, and/or perceived
avaricious intent of doctors and third party payers. Regardless of whether they were
insightful or not, participants characterized with worry complained about their healthcare
and/or focused on their symptoms. This ﬁnding parallels the results of an earlier
qualitative study of somatizing patients [159]. All worried participants shared stories of
achievement, action, and altruism. For at least one participant, the association of worry
with illness and health seeking behavior was unrecognized prior to this study. No worried
participant reported explicitly discussing his or her underlying fear with a healthcare
provider. At least one participant perceived a system and/or physician level banier to

such a discussion,

52

We must acknowledge important limitations. Our ﬁndings, like other qualitative
studies with small samples, may not be applicable to other primary care patients with
MUS and are subject to the biases of the investigators. Findings of associations are
preliminary. Secondly, relying on patient report, rather than direct observation may have
limited the content validity of some of our themes, especially those purported to describe
experiences and behaviors. However, the information obtained from interviews more
closely approximates the information available to clinicians from patients like those in
our study and therefore may be more useful clinically.

Moreover, this study has study generated several hypotheses for later quantitative
research: 1) Perceptions, like worry and entitlement, are more important than negative
experiences in the well-being and behavior of patients with MUS, 2) complaining about
healthcare and or a pervasive emphasis on symptoms is a marker for unrecognized on
unexpressed worry in some high utilizing primary care patients with MUS, 3)
systematically identifying and addressing concern about undiagnosed organic disease will
improve and/or alleviate symptoms focus and/or complaining behavior in some high

utilizing patients with MUS.

4.2 Conclusions

This descriptive study identiﬁed eleven themes that described the experiences,
perceptions, and behaviors of these high utilizing primary care patients with MUS.
Psychological insight was associated with achievement, action, and altruism. Conversely,
lack of insight was associated with entitlement and symptoms focus. Previously

unexpressed concern was associated with complaining and symptoms focus even in those

53

with insight, achievement, action, and altruism. These patterns of diverse personal
features underscore the potential value of unique diagnostic and treatment approaches in

similar high utilizing primary care patients with MUS.

4. 3 Practice implications

Our study provides support for emphasizing effective communication in treatment
of MUS [102]. Distrustful and complaining high utilizing patients are likely to beneﬁt
from empathic listening, investigation of the source of their distrust and education about
its relation to their problems. Recognizing and addressing concern about undiagnosed
organic disease may improve patient satisfaction and outcomes, and increase clinicians’
conﬁdence with their treatment. We also identiﬁed improved insight and patient
responsibility (opposite of “entitlement”) as possible worthwhile goals of treatment.
Characteristics and stories of achievement, positive action, and altruism may be helpﬁil in

motivating MUS patients to live lives that are more productive.

54

 

Table 5: Qualitative Study - Preliminary and Working Themes

 

Preliminary Themes Working Themes

 

 

l. Behavior/Action 1. Primary mechanism
(Pleasure in life, Coping / Dysfunction, Job satisfaction)
2. Primary relationships 2' Secondary gain
(Duration of marriage)

. ‘ . 3. Insight (mind/body
Secondary relationships connection)

3. Doctor-patient relationship 4 Emotionality

4. Mechanism of illness

ldentity/ invisible, Stage of development, Personality, Locus of
control, Number of siblings, Location of patient, Abuse,
Family History, Explanatory model 6. Fear of physical disease

5. Symptoms focus

5. Physical Symptoms

Fear of physical disease, Care seeking, Secondary gain 7' Quality Of dominant

relationships
6. Diagnosis
Medical (primary or secondary), Psychiatric diagnosis 8, Obesity
(primary or secondary), MUS diagnosis (minor acute,
somatization, neither)

7. Emotionality

Expression during interview, Evidence of emotionality in life,
Insight / psychological savvy)

8. Excessive testing / medicalization

9. Reaction to interview and study

10. Religion / spirituality

1 1. Healthcare system

12. Litigation

 

l3. Education/training

 

55

 

Table 6: Qualitative Study - Final Themes with Deﬁnitions (11 = 11)

Category Theme Participant ID
Experiences 1. Childhood trauma 3, 4, 6, 7, 8, 9, 13,
(All are Expressions of traumatic experiences at a young age 14’ 15’ 19
negative)

2. Adulthood abuse

Explicitly described physical, verbal or sexual 6’ 12’ 14’ 15’ 17’ 19

abuse during adulthood

3. Family patterns of distress and/or dysfunction

, . . .. 1.3.4.5,6,8,9,
Expressrons of illnesses, behavrors, or conditions 10, 11’ 13, 14, 15,

that were repeated among different family

. . 16, 17, 18, 19
members; also includes expressed negative
emotions about the actions and intentions of family
members and other personal relationships

Perceptions 4. Entitlement 1, 3, 4, 6, 10, 14,
a) Negative Inferred lack of participant’s sense of accountability 16’ '7’ 18
perceptions for his or her actions or inactions, usually from

statements that offer symptoms as excuses for not
being able to fulﬁll societal roles

5 . Worry

Expressed or inferred participant concern about 3’ 5’ 7’ 9’ 12’ 19

serious undiagnosed disease. Neither normal tests
nor doctors’ benign assessments of their symptoms
reassured patients who expressed worry. Either they
had personally experienced a medical error, or they
knew someone who had.

6. Insight
5, 7, 8,11,12,15

Expressed or inferred awareness of the relationship
between personal psychological stress and physical

b) Positive symptoms

perception

56

Table 6 Continued
Category

Behaviors

a) Negative
behaviors

b) Positive
behaviors

Theme

7. Symptoms focus

A pervasive emphasis on symptoms

8. Complaining

Expressed and inferred dissatisfaction with
healthcare system or providers.

9. Achievement

Expressions of higher education, supervisory role,
professional status, entrepreneurship, and/or
creative activities

10. Action

Expressed or inferred ability to cope effectively or
change behavior for the better

I I . Altruism

Spontaneous descriptions of volunteer activity,
signiﬁcant care-taking or meaningful work

57

Participant

1,3,4, 5, 6, 9, 10,
13, 14,17, 18,16,
19

3, 7,9,11,12,13,
14,16,19

2,3,5, 7, 8, 9, 10,
11, 12, 13, 14,15,
16,17, 18,19

2,3,5,7,8,9,11,
12, 13,15,16,19

3, 5, 7. 8, 9,11,12,
15,19

Appendix 1: Semi-structured Questionnaire for interviews

Introduction:
1. Myself
2. The study

3. The interview (audiotape, notes)

Pause for questions

Open-ended beginning (30 - 45 minutes):
Say: “Tell me about yourself”
Use patient-centered method (patient-directed, empathic) to expand the patient’s story of

the physical, personal and emotional aspects of their illness

Directive Questioning (30 - 45 minutes): (Ask these questions if the corresponding
topics have not been discussed, continue to expand newly raised topics with clarifying
questions and patient-centered techniques.)

Explanatory models:

1. What do you believe is the root cause of your problems?
2. What are your concerns about your problems?
3. How have your health problems affected your life?

Locus of control:

1. Who do you feel has the most control over your health (life)?
Health-seeking behavior:

2. Whom do you turn to for information about your health? Is there

anyone else?
Relationships:

3. How is your relationship with your healthcare provider

0 What are your expectations when you go to the doctor?

0 Do your issues get resolved to your satisfaction?

58

0 How satisﬁed are you with the care you have received?

Expectations for the future:
1. What are your expectations about your future?

“Added after Interview 2**: (Ask these questions if not already discussed)
9. Describe your childhood.

a. Have you ever been abused?

59

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