Cognitive Behavioral Therapy for the Treatment of Juvenile Fibromyalgia:

Objective—Juvenile fibromyalgia syndrome (FMS) is a chronic musculoskeletal pain disorder in children and adolescents for which there are no evidence-based treatments. The objective of this multisite, single-blind, randomized clinical trial was to test whether cognitive–behavioral therapy (CBT) was superior to fibromyalgia (FM) education in reducing functional disability, pain, and symptoms of depression in juvenile FMS. Methods—Participants were 114 adolescents (ages 11–18 years) with juvenile FMS. After receiving stable medications for 8 weeks, patients were randomized to either CBT or FM education and received 8 weekly individual sessions with a therapist and 2 booster sessions. Assessments were conducted at baseline, immediately following the 8-week treatment phase, and at 6-month followup. Results—The majority of patients (87.7%) completed the trial per protocol. Intent-to-treat analyses showed that patients in both groups had significant reductions in functional disability, pain, and symptoms of depression at the end of the study, and CBT was significantly superior to FM education in reducing the primary outcome of functional disability (mean baseline to end-of© 2012, American College of Rheumatology Address correspondence to Susmita Kashikar-Zuck, PhD, Division of Behavioral Medicine and Clinical Psychology, MLC 3015, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229. Susmita.Kashikar-Zuck@cchmc.org. AUTHOR CONTRIBUTIONS All authors were involved in drafting the article or revising it critically for important intellectual content, and all authors approved the final version to be published. Dr. Kashikar-Zuck had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Study conception and design. Kashikar-Zuck, Arnold, Bean, Powers, Graham, Schikler, Banez, Lovell. Acquisition of data. Kashikar-Zuck, Ting, Graham, Passo, Schikler, Hashkes, Spalding, Lynch-Jordan, Banez, Richards, Lovell. Analysis and interpretation of data. Kashikar-Zuck, Arnold, Bean, Powers, Banez, Lovell. NIH Public Access Author Manuscript Arthritis Rheum. Author manuscript; available in PMC 2013 June 24. Published in final edited form as: Arthritis Rheum. 2012 January ; 64(1): 297–305. doi:10.1002/art.30644. N IH PA Athor M anscript N IH PA Athor M anscript N IH PA Athor M anscript treatment difference between groups 5.39 [95% confidence interval 1.57, 9.22]). Reduction in symptoms of depression was clinically significant for both groups, with mean scores in the range of normal/nondepressed by the end of the study. Reduction in pain was not clinically significant for either group (<30% decrease in pain). There were no study-related adverse events. Conclusion—In this controlled trial, CBT was found to be a safe and effective treatment for reducing functional disability and symptoms of depression in adolescents with juvenile FMS. Juvenile fibromyalgia syndrome (FMS) is a chronic pain condition that is estimated to affect 2–7% of school-age children (1–3), primarily adolescent girls. As in adult fibromyalgia (FM), juvenile FMS is characterized by persistent and widespread musculoskeletal pain, sleep difficulty, fatigue, and mood disturbances (3). Patients with juvenile FMS experience substantial impairment in physical, school, social, and emotional functioning (4–6), and the majority of them continue to have ongoing symptoms and functional disability into late adolescence and early adulthood (7). Although progress has been made in the understanding and treatment of adult FM (8), there are few studies of juvenile FMS. Two recent metaanalytic reviews of cognitive–behavioral therapy (CBT) clinical trials for FM arrived at slightly different conclusions, with one supporting the effectiveness of CBT (9) and the other reporting no significant effect on pain and function (10). However, both reviews indicated significant improvement in coping and emphasized the need for higher quality trials of CBT. It is unclear whether findings from the 2 meta-analytic reviews, based mainly on adult FM patients, are applicable to pediatric patients. In a recent meta-analysis of psychological therapies for pediatric chronic pain (mostly headache and abdominal pain), Palermo et al (11) reported a large positive effect for pain reduction. It is possible that behavioral interventions found to be promising for adults with FM can be even more effective in juvenile FMS because they are implemented at an early age when pain coping and lifestyle factors are more malleable. There are no rigorously controlled studies of CBT for juvenile FMS at present. Early evidence for possible efficacy of CBT for improving function and decreasing pain and symptoms of depression in children and adolescents with juvenile FMS was shown in 2 small-scale studies (12,13). Unfortunately, neither study controlled for the nonspecific therapeutic effects of attention and support from health professionals, which by themselves could have resulted in improved outcomes. Therefore, the specific effects of CBT that go beyond providing emotional support and include intensive training in behavioral pain coping skills targeted at improving functioning by increasing patients' psychological coping and confidence in pain self-management skills have not yet been tested in a controlled study. We performed a multisite, randomized controlled trial in a sample of children and adolescents with juvenile FMS to rigorously assess the safety and efficacy of an 8-session, individually delivered CBT intervention compared to an 8-session supportive FM education program in reducing functional disability, pain severity, and depressive symptoms. The CBT treatment protocol was developmentally tailored for older school-age children and adolescents and included parental instruction to ensure family support for the behavioral interventions. We hypothesized that 1) CBT would be significantly more effective than FM education in reducing functional disability (primary outcome) in youth with juvenile FMS and 2) CBT would be significantly more effective than FM education in reducing pain severity and symptoms of depression (secondary outcomes). Unlike a “no-treatment” placebo control, the FM education condition was designed to be an “attention control” condition in which the nonspecific psychological benefits of the therapeutic relationship and educational information provided were expected to lead to some improvement in the short term (posttreatment assessment). However, we hypothesized that CBT would lead to sustained benefit and be demonstrably superior to FM education in reducing disability, pain, Kashikar-Zuck et al. Page 2 Arthritis Rheum. Author manuscript; available in PMC 2013 June 24. N IH PA Athor M anscript N IH PA Athor M anscript N IH PA Athor M anscript and symptoms of depression 6 months after the end of active treatment (end-of-study assessment). Selection of the outcome domains of function, pain severity, and symptoms of depression was consistent with the Pediatric Initiative on Methods, Measurement, and Pain Assessment in Clinical Trials (PedIMMPACT) guidelines for measurement of outcomes in clinical trials of pediatric chronic pain (14). Functional disability was selected as the primary outcome because the goal of CBT is to reduce pain-related disability by teaching patients both coping and self-management behaviors (and providing parental guidance in behavior management) to improve function, while secondarily reducing pain severity and symptoms of depression. Other outcomes of interest such as tender point sensitivity, health-related quality of life (HRQOL), sleep quality, and physician's global assessment were also measured at each assessment. PATIENTS AND METHODS The study was conducted at 4 pediatric rheumatology centers in the Midwestern US. The enrollment period was between December 2005 and December 2009. Treatment and followup of all patients were completed in June 2010. Children between ages 11 and 18 years were eligible if they 1) met juvenile FMS classification criteria as determined by a pediatric rheumatologist (3), 2) were receiving stable medications for 8 weeks and were willing to continue receiving stable medications for the duration of the study, 3) reported average pain severity ≥4 on a 0–10-cm visual analog scale (VAS) based on 1 week of daily pain diaries, and 4) obtained a score >7 on the Functional Disability Inventory (FDI) (15), indicating at least mild disruption in daily activities due to juvenile FMS symptoms. Exclusion criteria were 1) other rheumatic disease, such as juvenile arthritis or lupus; 2) documented developmental delay; 3) current panic disorder or major depression, or lifetime bipolar disorder or psychosis; and 4) use of opioids. The study protocol was approved by the Institutional Review Boards at each of the 4 sites. Written informed consent was obtained from parents of the patients, and written assent was obtained from the patients. An independent Data and Safety Monitoring Board assembled by the study sponsor (the National Institutes of Health) and composed of an adult rheumatologist, pediatric rheumatologist, pediatric psychologist, biostatistician, physician medical/safety monitor, and bioethicist monitored the conduct of the trial.