Engagement in Cognitive-Behavioral Therapy for Chronic Pain Management Is Associated with Reductions in Healthcare Utilization in Pediatric Sickle Cell Disease
Authors: Sil Soumitri , Lai Kristina , Lee Jennifer L. , Gilleland-Marchak Jordan , Thompson Beth , Cohen Lindsey L , Lane Peter A. , Dampier Carlton 
Published: February 2020

Introduction: Chronic pain in sickle cell disease (SCD) is a multifactorial complication that can contribute to high healthcare utilization. Multidisciplinary treatments going beyond medication alone are needed for the most effective chronic pain management. Cognitive-behavioral therapy (CBT) is effective for youth with chronic pain and focuses on improving daily functioning and coping, but the clinical effectiveness for chronic SCD pain has not been evaluated. This study examined changes in healthcare use over time for youth with chronic SCD pain who participated in CBT compared to controls with chronic SCD pain who never initiated CBT.
Methods: Youth receiving care at comprehensive SCD clinics at three tertiary care locations at Children's Healthcare of Atlanta were included if they were aged 6-18 years, any SCD genotype, and referred to a pediatric psychology outpatient clinic for chronic pain management from November 2014-March 2018. Youth were excluded if they received bone marrow transplantation during the study period, had ongoing CBT past the study period, or were not actively followed for ≥1 year of medical care pre- or post-CBT. Patients were grouped based on therapy attendance: Established Care (i.e., attended ≥3 CBT sessions within 3 consecutive months); Early Termination (i.e., attended &lt;3 CBT sessions within 3 consecutive months); or Control (i.e., did not attend any CBT visits). Patient-reported outcomes included typical pain intensity, functional disability, and coping efficacy at pre- and post-treatment. Healthcare utilization outcomes were abstracted from electronic medical records including: number of inpatient admissions for pain, total inpatient days for pain, and emergency department dependency ratio (EDR; ratio of ED visits to sum of ED and outpatient visits). For the treatment groups, utilization outcomes were calculated from 12-months prior to the first CBT visit, and from 12-months following the last CBT visit. For the control group, outcomes were calculated for 12-months prior to the referral date, and from 12-months following the average duration of CBT for treatment groups (i.e., 3.5 months) to account for passage of time. Changes over time in inpatient admissions, hospital days, and EDR were evaluated separately using linear mixed effect models with a random effect for person-specific intercepts and slopes, which were retained based on model contribution determined by Bayesian Information Criterion. Time, patient characteristics, SCD-modifying treatments, therapy attendance, number of CBT sessions, and interaction effects were initially included in the models; the most parsimonious models were chosen based on backward selection.
Results: At time of referral, youth (n=101) were on average (M) 13.4 years old (SD=2.92), 56.4% female, 68.1% HbSS or HbSβ0, 63.9% prescribed hydroxyurea, and 12.6% received chronic transfusions. The Control (n=44) and Treatment Groups (n=57) did not significantly differ by age, sex, genotype, or treatment with hydroxyurea or chronic transfusion. Based on therapy attendance, 36.1% Established Care, 21.8% were Early Termination, and 42% Controls. Adjusting for age, genotype, and hydroxyurea, patients who terminated CBT early had increased admissions and total hospital days over time compared to controls; those who established care had a reduction in admissions and hospital days over time compared to controls (F's=3.27-3.61, p's&lt;.05). EDR decreased by 0.1 over time for Established Care; for every 1 completed CBT session, EDR was further reduced by 0.01 (p&lt;.05). Patients who completed CBT (n=18) reported decreases in typical pain intensity (Mpre= 5.47, SD=2.24; Mpost=3.76, SD=2.84; p&lt;.01), functional disability (Mpre=26.24, SD=8.45; Mpost=15.18, SD=10.85; p&lt;.001), and improved coping efficacy (Mpre=8.0, SD=2.21; Mpost=9.65, SD=2.94; p&lt;.05) from pre- to post-treatment.
Conclusions: Establishing care in CBT may support reductions in admissions for pain, length of stay, and ED dependency for youth with chronic SCD pain beyond the potential effects of age, genotype, and SCD-modifying treatments. Reductions in utilization may be partially supported by patient-reported improvements in functioning, coping, and lower pain intensity following CBT. Reducing barriers to access and enhancing clinical implementation of multidisciplinary treatments may optimize the health of youth with chronic SCD pain.

Disclosures
Lane: NHLBI: Research Funding; CDC: Research Funding; GA Dept: Other: Contract for newborn screeninjg follow-up services services; Bio Products Laboratory: Other: Sickle Cell Advisory Board; FORMA Therapeutics: Other: Clinical Advisory Board. Dampier:Pfizer: Consultancy, Membership on an entity's Board of Directors or advisory committees, Research Funding; Micelle BioPharma: Consultancy, Membership on an entity's Board of Directors or advisory committees, Research Funding; Global Blood Therapeutics: Consultancy; Epizyme: Consultancy; Novartis: Consultancy, Membership on an entity's Board of Directors or advisory committees, Research Funding; Katz Foundation: Research Funding; Modus Therapeutics: Consultancy; NIH: Research Funding; Merck: Research Funding.