jurnal somatoform oxford

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Somatic symptoms are common in children but diagnosable conditions are rare. In a community sample of 540 school-age children, Garber, Walker, and Zeman (1991) found that only 1.1% of children met full diagnostic criteria for somatization disorder according to DSM-III-R criteria. Similarly, the esti- mated rate of conversion disorders is described as less than 1% of the general population (APA, 1994). Although there have been no controlled treat- ment outcome studies of somatoform disorder in children, case reports and case series have been pub- lished that describe treatment of such problems as persistent somatoform pain disorder (e.g., Lock & Giammona, 1999) and conversion reactions (e.g., Donohue, Thevenin, & Runyon, 1997; Woodbury, Journal of Pediatric Psychology, Vol. 26, No. 7, 2001, pp. 429–434 Treatment of Functional Impairment in Severe Somatoform Pain Disorder: A Case Example Tonya Mizell Palermo, PhD, and Mark S. Scher, MD Case Western Reserve University School of Medicine Objective: To describe the assessment and treatment of severe functional impairment in a young female ad- olescent with somatoform pain disorder. Methods: Treatment included an inpatient hospital admission using a rehabilitation approach and behavior modification program. Standardized assessment of functional impairment and health-related quality of life was performed at baseline and follow-up. Diagnostic evaluation and treatment costs were computed using insurance and hospital billing data. Results: Pre-/postintervention measures of functional disability indicated significant improvement in physi- cal and psychosocial functioning in everyday activities. Although costly, the inpatient admission decreased frequent health care use over the short term. Conclusions: Delay in diagnosis of somatoform disorders may seriously extend children’s disability and re- quire more intensive treatment. Functional disability is a critical measure of treatment outcome in children with severe somatoform disorder. Future research concerning interventions for children with a broad range of recurrent and chronic pain symptoms can be strengthened through a focus on reducing functional dis- ability. Key words: somatoform disorder; children; functional impairment; pain. Somatoform disorders are described in DSM-IV (American Psychiatric Association [APA], 1994) as the presence of physical symptoms suggesting an underlying medical condition, but the medical con- dition is neither found nor fully accounts for the level of functional impairment. DSM diagnoses in- clude somatization disorder, conversion disorder, pain disorder, hypochondriasis, and body dysmor- phic disorder, with related conditions including vo- cal cord dysfunction, reflex sympathetic dystrophy, and recurrent abdominal pain. 2001 Society of Pediatric Psychology All correspondence should be sent to Tonya Palermo, Division of Behav- ioral Pediatrics and Psychology, Rainbow Babies & Children’s Hospital, 11100 Euclid Ave., Cleveland, Ohio 44106-6038. E-mail: txm36@po. cwru.edu.

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Somatic symptoms are common in children butdiagnosable conditions are rare. In a communitysample of 540 school-age children, Garber, Walker,and Zeman (1991) found that only 1.1% of childrenmet full diagnostic criteria for somatization disorderaccording to DSM-III-R criteria. Similarly, the esti-mated rate of conversion disorders is described asless than 1% of the general population (APA, 1994).

Although there have been no controlled treat-ment outcome studies of somatoform disorder inchildren, case reports and case series have been pub-lished that describe treatment of such problems aspersistent somatoform pain disorder (e.g., Lock &Giammona, 1999) and conversion reactions (e.g.,Donohue, Thevenin, & Runyon, 1997; Woodbury,

Journal of Pediatric Psychology, Vol. 26, No. 7, 2001, pp. 429–434

Treatment of Functional Impairment in SevereSomatoform Pain Disorder: A Case Example

Tonya Mizell Palermo, PhD, and Mark S. Scher, MDCase Western Reserve University School of Medicine

Objective: To describe the assessment and treatment of severe functional impairment in a young female ad-

olescent with somatoform pain disorder.

Methods: Treatment included an inpatient hospital admission using a rehabilitation approach and behavior

modification program. Standardized assessment of functional impairment and health-related quality of life

was performed at baseline and follow-up. Diagnostic evaluation and treatment costs were computed using

insurance and hospital billing data.

Results: Pre-/postintervention measures of functional disability indicated significant improvement in physi-

cal and psychosocial functioning in everyday activities. Although costly, the inpatient admission decreased

frequent health care use over the short term.

Conclusions: Delay in diagnosis of somatoform disorders may seriously extend children’s disability and re-

quire more intensive treatment. Functional disability is a critical measure of treatment outcome in children

with severe somatoform disorder. Future research concerning interventions for children with a broad range

of recurrent and chronic pain symptoms can be strengthened through a focus on reducing functional dis-

ability.

Key words: somatoform disorder; children; functional impairment; pain.

Somatoform disorders are described in DSM-IV(American Psychiatric Association [APA], 1994) asthe presence of physical symptoms suggesting anunderlying medical condition, but the medical con-dition is neither found nor fully accounts for thelevel of functional impairment. DSM diagnoses in-clude somatization disorder, conversion disorder,pain disorder, hypochondriasis, and body dysmor-phic disorder, with related conditions including vo-cal cord dysfunction, reflex sympathetic dystrophy,and recurrent abdominal pain.

� 2001 Society of Pediatric Psychology

All correspondence should be sent to Tonya Palermo, Division of Behav-ioral Pediatrics and Psychology, Rainbow Babies & Children’s Hospital,11100 Euclid Ave., Cleveland, Ohio 44106-6038. E-mail: [email protected].

DeMaso, & Goldman, 1992). Treatments successfulin remediating symptoms and improving children’sfunctioning include rehabilitation approaches (e.g.,Brazier & Venning, 1997), behavioral techniques(e.g., Campo & Negrini, 2000), relaxation tech-niques, and family therapy (e.g., Lock & Giam-mona, 1999).

Measuring Functional Impairment inChildren With Severe Somatization

The key feature in most definitions of clinically sig-nificant somatization is the excessive functional im-pairment that results from the patient’s somaticcomplaints (Kellner, 1991). The majority of case re-ports of somatoform disorder have documentedtreatment efficacy through patient or parent reportof physical functioning such as school attendanceand activity participation (e.g., Brazier & Venning,1997; Campo & Negrini, 2000). Follow-up is oftenlimited to the immediate posttreatment period.

The methodological contribution of this casestudy is twofold. One contribution is to demon-strate the applicability of a standardized written in-strument to measure child functional disabilitybefore and after treatment. The second contributionis an extended follow-up period (6 months) to dem-onstrate treatment effects over time.

Purpose of the Case Report

The purposes of this case report of severe somato-form pain disorder in a young adolescent are (1) toillustrate the use of standardized outcome measuresof functional disability, (2) to demonstrate efficacyof rehabilitation modeling and behavioral interven-tion in increasing function, and (3) to present re-search implications for treatment of functionaldisability in a broad range of patients with chronicpain symptoms not fully explained by organic pa-thology. A related goal of the article is to report dataon the cost-benefit ratio of diagnosis and treatmentof a pediatric patient with severe somatoform paindisorder. Presumably, children with somatoformdisorder who fail to be successfully treated couldcontinue to have bouts of symptoms into adult-hood (e.g., Walker, Guite, Duke, Barnard, & Greene,1998) that result in inappropriate medical utiliza-tion, interference with occupational attainment,and significant costs to society.

Method

History and Diagnostic Work-Up

The patient, an 11-year-old Caucasian girl, pre-sented with a 5-month history of whole body painthat eventually lead to paralysis of her extremities.She had become nonambulatory and totally depen-dent on her parents for all self-care activities overthe prior 2 months. Because of the extensive immo-bility, there were concerns about progressive musclewasting and contractures.

The onset of symptoms followed a documentedviral infection (with evidence of Epstein-Barr viralinfection). The diagnostic work-up for this patientinvolved pediatric office visits and subspecialty pe-diatric evaluation by immunology, neurology, rheu-matology, and physical therapy. Results of ordereddiagnostic procedures (e.g., blood work, MRI) werenegative. Because the patient’s functioning contin-ued to deteriorate, a second neurologist was con-sulted who recommended an acute psychiatrichospitalization. After 3 days of medical and psychi-atric evaluation, the patient was discharged fromthe psychiatric facility with a diagnosis of severe fi-bromyalgia. Discharge recommendations includedweekly counseling with a licensed social worker andevaluation at an adult pain center. During this time,the patient’s functioning had deteriorated to com-plete immobility and she was using a wheelchair.The family was directed to a third pediatric neurolo-gist (MS), who confirmed a somatoform disorderand recommended treatment with an inpatient pe-diatric admission.

Inpatient Admission

Setting. The patient was admitted to the collabora-tive care unit (CCU) at a tertiary care children’s hos-pital with a diagnosis of severe somatoform paindisorder. The CCU is a subacute medical unit staffedby a medical director, a developmental and rehabili-tative pediatrics specialist, a consulting neurologist,nursing staff, a dietitian, social worker, teachers,and respiratory, occupational, speech, and physicaltherapists. Psychology provides consultation on areferral basis.

History and Observations. The patient lived withher mother, father, and three sisters. She attendedthe fifth grade in a small religious school and wasreported to be an above average student. She had

430 Palermo and Scher

Health-Related Quality of Life (HRQOL). The ChildHealth Questionnaire (CHQ-PF50; Landgraf, Ab-etz, & Ware, 1996) is a 50-item measure that assesseschildren’s quality of life (e.g., physical functioning,limitations in schoolwork and activities withfriends due to physical health) and measures theemotional impact of children’s health on the care-giver respondent (e.g., the degree he or she feelslimited in personal time due to the child’s health).Severe limitations were noted in many areas, in-cluding the patient’s ability to perform all physicalactivities and her ability to do schoolwork and ac-tivities with friends. The patient was also reportedto have extremely severe, frequent, and limitingbodily pain and to have low self-esteem. The impactof the patient’s health on caregiver emotional dis-tress and personal time was also perceived to bequite limiting.

Treatment

The patient was hospitalized for 22 days in theCCU. The primary intervention strategies used dur-ing the admission were staff education, rehabilita-tion modeling, and operant behavioral techniques.Treatment was modeled after published case de-scriptions provided by Brazier and Venning (1997)and Gooch, Wolcott, and Speed (1997).

The consulting psychologist (TMP), who coordi-nated and developed the behavioral program forthe patient, conducted staff education focused onmethods of communication with the patient abouther pain and symptoms and use of contingent rein-forcement strategies for well behavior. An article de-scribing a rehabilitation approach to treatment ofsomatoform disorders (Brazier & Venning, 1997)was provided to all staff.

Approach to treatment focused on an interpre-

stopped attending school entirely several monthsbefore the hospital admission, with her parents pro-viding tutoring at home. Parents were both em-ployed full-time outside of the home. Familyhistory was significant for maternal depression, andanother sibling had a diagnosis of bipolar disorderand ulcerative colitis.

Several psychosocial stressors had been identi-fied in the months preceding the patient’s illness.The patient’s sister reportedly experienced sexualabuse by a family friend (that had been recently re-vealed), and the patient experienced a tremendouspersonal athletic failure a month before the onsetof symptoms.

Upon admission to the CCU, the patient dem-onstrated constant grimacing, grunting, and hold-ing her breath in pain. She was immobile in bedexcept for vocalizing and moving her eyes. Her self-report of pain indicated severe pain intensity (10 of10) in her toes, lower legs, thighs, hip, stomach,chest, head, shoulders, elbows, fingers, and thumbs.The patient was cooperative and polite to hospitalstaff and did not appear overly troubled by her symp-toms. When asked direct questions, she stoppedgrunting and answered in a normal tone, even jok-ing at times. She was receptive to the hospital ad-mission because she felt that “someone finally be-lieved that she needed medical help.” The patient’sparents and siblings showed emotional concernwhen the patient expressed pain and treated the pa-tient as totally dependent on others for assistance.

Procedures: Baseline Assessment

Anxiety and Depression. The patient denied any sig-nificant anxiety symptoms (Multidimensional Anx-iety Scale for Children [MASC]; March, 1997) ordepressive symptoms (Child Depression Inventory[CDI]; Kovacs, 1992).

Functional Disability. The Functional DisabilityInventory (FDI; Walker & Greene, 1991) was com-pleted to describe the extent of restriction in per-forming 15 standard activities (e.g., walking to thebathroom, being at school all day, getting to sleepat night). Response categories range from “notrouble” to “impossible” to perform each activity.Scores range from 0 to 60, with higher scores indi-cating more disability. As seen in Figure 1, the pa-tient’s self-report and parental report indicatedsignificant difficulty performing most activities atbaseline (total score � 53 and 49, respectively).

Severe Somatoform Pain Disorder 431

Figure 1. Functional disability scores at baseline, 1 month post-

discharge, and 6 months postdischarge.

tation of the patient’s symptoms as physical, butamenable to full recovery through a gradual in-crease in function with physical and occupationaltherapies. Rehabilitation was coupled with contin-gent reinforcement for improvement in functionand withdrawal of reinforcement for continuingsigns of disability. Emphasis was placed on strength-ening muscles and letting her pain go away of itsown accord. A behavioral level system was usedwith the patient to provide daily contingencies forher function and achievement of daily therapygoals.

The patient participated in physical, occupa-tional, and speech therapy and hospital school tu-toring during her admission. Her schedule followeda standard school day with therapies between 7:30a.m. and 4:00 p.m. She earned points on a daily goalcard for each therapy and self-care activity com-pleted during the day (e.g., toileting, feeding).These points resulted in different levels of privilegeseach day, ranging from full privileges (in which thepatient could do hospital activities and see visitorsin the evenings) to in-room privileges with no vis-itors.

Results

Symptom Reduction and Improvementin Function

Therapy goals set by physical, occupational, andspeech therapists were to increase mobility and self-care, decrease pain, and reduce grunting and pitchbreaks. The patient made daily progress in increas-ing movement in her extremities and performingself-care activities. Her upper extremity mobility re-turned first and led to independent feeding. A corre-sponding reduction in pain intensity was reported.The patient achieved goals of using normal voicequality without grunting and holding her breath.On hospital day 19, the patient ambulated indepen-dently with a walker. By hospital day 21, she wasambulating independently without assistive de-vices. She was discharged on day 22 because she hadreached treatment goals of independent walking,self-care, and feeding.

Follow-Up

Short-Term Follow-Up. The patient returned to schoolon the day after hospital discharge and attended

daily for the remaining 3 weeks of the school year.She began to participate in some physical activitiesat recess. At 1 month postdischarge, the FDI andCHQ were again administered to the patient andher parents to obtain standardized outcome data onher functional disability and quality of life. As seenin Figure 1, the patient and her mother reportedmarked improvement across all activities, with only“running” identified as “impossible.” CHQ re-sponses revealed overall improvement in all do-mains of physical and psychosocial health. Somecontinuing areas of difficulty were reported as mod-erate limitations in the area of social roles (e.g.,school and peer activities limited due to emotionaland behavioral difficulties), family activities, andemotional distress for caregivers. Physical therapyreevaluation indicated that the patient hadachieved normal upper extremity strength, normalbalance, and showed only mild lower extremityweakness. She met physical therapy goals of beingable to walk on her toes, shift weight on a balanceboard, and jump. Pain complaints reported by thepatient were limited to challenging physical activi-ties (e.g., walking on toes, jumping).

Six-Month Follow-Up. A 6-month postdischargephone interview was conducted. The patient was re-ported to have continued to make gains in physicalfunctioning and was attending school consistently(only two missed school days due to illness) andperforming all physical activities including run-ning, jumping, and bike riding. On the FDI, the pa-tient and her mother (see Figure 1) reported veryfew limitations and that no activities were “impos-sible.” Teacher data confirmed the patient’s partici-pation in gym class and physical activities at recess.The patient reported no pain or somatic com-plaints.

Cost Analysis

During the 5-month period of extended diagnosisand treatment prior to the inpatient admission, thepatient incurred $16,314 in medical charges. Thesecharges were for medical provider visits ($12,170),diagnostic procedures (e.g., CT scan, MRI), andmedications ($4,144). The patient’s 22-day inpa-tient admission to the CCU totaled $29,163, includ-ing her hospital room; medical supplies; physical,occupational, and speech therapy; and psychologi-cal services. The patient was covered by private in-surance with a mental health carve out.

In the 6 months postdischarge, medical costs in-

432 Palermo and Scher

mation used in conjunction with detailed behav-ioral observation.

Because functional impairment may occur withor without known organic disease, descriptionsof interventions to improve functioning in chil-dren with medically unexplained symptoms haveincreased relevance in the pediatric psychologytreatment literature. For example, interventionstrategies used to decrease functional impairment inchildren with severe somatization may contributeto treatments to lessen the functional impact ofpain in children with more common persistentpains (e.g., headaches, recurrent abdominal pain).In fact, the literature on treatment of recurrent andchronic pain in children is limited by the lack ofcontrolled treatment studies (Walco, Sterling,Conte, & Engel, 1999) and the almost exclusive fo-cus on symptom relief to the neglect of functionalimprovement (Palermo, 2000).

This case report illustrated the diagnostic uncer-tainty that somatization can create among pediatricmedical providers, as well as the profound impair-ment and disability that may result from unsuccess-ful treatment. This combination of factors results incostly treatment approaches for somatoform dis-order. However, the economic costs of severe soma-tization are tremendous, affecting unnecessaryhealth care costs for medical provider visits, diag-nostic procedures, and medications, as well as a sig-nificant loss of family and patient productivity.Cost data illustrated that untreated somatoform dis-order in children can have a major economic im-pact, but with appropriate treatment, potentiallifelong patterns of unnecessary health care costsmay be avoided.

This study is limited by the single case pre-posttreatment design. A multiple-baseline treatment de-sign would have most effectively demonstrated theefficacy of rehabilitation modeling and behavioraltreatment. However, a single case design can con-tribute important ideas for future research on treat-ment of somatoform disorders in children. As thiscase illustrated, future studies are needed to clarifythe role of individual and family factors in the de-velopment or maintenance of somatization andfunctional impairment so that more focused inter-ventions may be targeted to specific children andfamilies.

Received August 1, 2000; revisions received December 7,2000; accepted January 30, 2001

cluded primarily outpatient physical therapy andpsychotherapy. Physical therapy services were pro-vided for the first 2 months (11 visits). The patienthad one follow-up visit with her neurologist. A pe-diatrician visit was made following a bike accident.The patient participated in ongoing psychotherapyon a weekly basis. Total costs in the 6 months post-discharge were estimated at $3,980.

These cost data indicate that severe somatoformpain disorder can be quite costly to diagnose andtreat. Essentially, the cost savings of successful treat-ment will be realized during the second year for thispatient. However, this investment of treatment in-terrupted the pattern of excessive medical use andshould return profound savings in medical costs ifa lifelong pattern of inappropriate health care useis altered.

Discussion

Somatoform disorder in children and adolescentscan present diagnostic and treatment challenges toa range of professionals caring for these children,including primary care pediatricians, subspecialtypediatric providers, psychiatrists, physical thera-pists, and psychologists (Garralda, 1999). Findingsfrom this case lend further support for the effective-ness of rehabilitation modeling and behavioralintervention in treatment of severe somatoformdisorder in children (Brazier & Venning, 1997;Gooch et al., 1997). The patient described in thiscase report presented with a history of many indi-vidual factors identified in the development of so-matization, such as family health problems andpsychological distress (Walker & Greene, 1989),high achievement (e.g., Dvonch, Bunch, & Siegler,1991), stressful life events, minor physical illnesses(Garralda, 1996), and inadequate medical advice oruncertainty of diagnosis.

This case report contributes to the literature onsomatoform pain disorder by highlighting the stan-dardized assessment of functional disability overtime. Standardized assessment of functional disabil-ity in children with severe somatization will helpto describe the patient population and presentingsymptoms, to compare treatment outcomes be-tween various studies, and to identify children whocontinue to demonstrate subtle functional impair-ment after treatment. Whereas written question-naires such as the FDI are cost-effective and easy toimplement, they will provide the most useful infor-

Severe Somatoform Pain Disorder 433

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