Non-Pharmacologic Intensive Interdisciplinary Pain Treatment in Pediatrics: Impact on Symptoms, Daily Functioning, and the Family Unit.

Objectives: To assess non-pharmacologic treatment outcomes pertaining to health-related quality of life (HRQoL) in youth with chronic idiopathic pain and their families. Methods: We conducted a retrospective cohort study of 115 youth with chronic idiopathic pain enrolled in a non-pharmacologic, hospital-based intensive interdisciplinary pain treatment (IIPT) program. HRQoL measures for the patient (Pediatric Quality of Life Inventory [PedsQL] short form) and family unit (PedsQL Family Impact) were collected on admission and discharge as part of routine clinical care. Changes in PedsQL scores were calculated using the Wilcoxon signed-rank test. Multivariable linear regression was used to explore factors associated with patient-level HRQoL. Results: Both individuals and the family unit reported that their HRQoL improved in all domains by program completion. Improvements in pain and allodynia were present for program participants at the time of completion as well as at the 3-month follow-up, suggesting durability of these effects. Conclusions: A non-pharmacologic IIPT program is a compelling treatment for pediatric and adolescent chronic idiopathic pain, for both patients and the family unit. Patients participating in this program had positive treatment outcomes with significantly improved subjective and objective measures of physical, emotional, social, and cognitive function.

Pediatric Complex Regional Pain Syndrome With and Without a History of Prior Physical Trauma at Onset.

OBJECTIVE: To determine whether differences exist between children with complex regional pain syndrome (CRPS) who identify an inciting physical traumatic event (group T) versus those without such history (group NT). METHODS: We performed a single-center, retrospective study of children diagnosed with CRPS, 18 years old or younger, presenting between April 2008 and March 2021 and enrolled in a patient registry. Abstracted data included clinical characteristics, pain symptoms, Functional Disability Inventory, psychological history, and Pain Catastrophizing scale for children. Charts were reviewed for outcome data. RESULTS: We identified 301 children with CRPS, 95 (64%) reported prior physical trauma. There was no difference between the groups regarding age, sex, duration, pain level, function, psychological symptoms, and scores on the Pain Catastrophizing Scale for Children. However, those in group T were more likely to have had a cast (43% vs 23%, P < 0.001). Those in group T were less likely to experience complete resolution of symptoms (64% vs 76%, P = 0.036). There were no other outcome differences between the groups. DISCUSSION: We found minimal differences in children with CRPS who report a prior history of physical trauma to those who do not. Physical trauma may not play as significant a role as immobility, such as casting. The groups mostly had similar psychological backgrounds and outcomes.

Improving Outcomes of Pediatric Lupus Care Delivery With Provider Goal-Setting Activities and Multidisciplinary Care Models.

OBJECTIVE: The present study was undertaken to evaluate high-quality care delivery in the context of provider goal-setting activities and a multidisciplinary care model using an electronic health record (EHR)-enabled pediatric lupus registry. We then determined associations between care quality and prednisone use among youth with systemic lupus erythematosus (SLE). METHODS: We implemented standardized EHR documentation tools to autopopulate a SLE registry. We compared pediatric Lupus Care Index (pLCI) performance (range 0.0-1.0; 1.0 representing perfect metric adherence) and timely follow-up 1) before versus during provider goal-setting activities and population management, and 2) in a multidisciplinary lupus nephritis versus rheumatology clinic. We estimated associations between pLCI and subsequent prednisone use adjusted for time, current medication, disease activity, clinical features, and social determinants of health. RESULTS: We analyzed 830 visits by 110 patients (median 7 visits per patient [interquartile range 4-10]) over 3.5 years. The provider-directed activity was associated with improved pLCI performance (adjusted ß 0.05 [95% confidence interval (95% CI) 0.01, 0.09]; mean 0.74 versus 0.69). Patients with nephritis in multidisciplinary clinic had higher pLCI scores (adjusted ß 0.06 [95% CI 0.02, 0.10]) and likelihood of timely follow-up than those in rheumatology (adjusted relative risk [RR] 1.27 [95% CI 1.02, 1.57]). A pLCI score of ≥0.50 was associated with 0.72-fold lower adjusted risk of subsequent prednisone use (95% CI 0.53, 0.93). Minoritized race, public insurance, and living in areas with greater social vulnerability were not associated with reduced care quality or follow-up, but public insurance was associated with higher risk of prednisone use. CONCLUSION: Greater attention to quality metrics is associated with better outcomes in childhood SLE. Multidisciplinary care models with population management may additionally facilitate equitable care delivery.

Development and evaluation of the patient-reported outcomes measurement information system (PROMIS) pediatric pain quality item bank and short form.

ABSTRACT: Pain is a common problem among children, particularly those with pediatric chronic diseases. Multifaceted assessment of pain can improve communication about pain and help clinicians characterize, differentiate, and treat a patient's unique experience of pain. Pain quality is an important domain of pain, describing the subjective sensory experiences associated with pain as well as the affective experiences of pain. The aim of the current study was to quantitatively evaluate the measurement properties of the 59 Patient-Reported Outcomes Measurement Information System pediatric pain quality candidate items developed as part of the National Institutes of Health's Patient-Reported Outcomes Measurement Information System initiative with input from children and adolescents with chronic pain. Participants included N = 448 pediatric patients between 8 and 18 years of age with chronic health conditions with a prominent component of chronic or recurrent pain, including juvenile fibromyalgia, juvenile idiopathic arthritis, and sickle cell disease. A confirmatory factor analysis revealed a unidimensional model fit the data best, with 56 of the 59 items demonstrating good psychometric properties for inclusion in the final measure. In addition, a consensus-building method was used to establish 2 versions of a short form measure-one with 8 items focused primarily on the sensory pain qualities and one with 8 items focused on affective pain qualities. The final measure shows good reliability and validity, and is recommended for use in research and clinical care with pediatric populations.

The Feasibility and Acceptability of Resilience Coaching for Adolescent Chronic Musculoskeletal Pain: A Single-Arm Pilot Trial.

Promoting Resilience in Stress Management (PRISM) is a well-established resilience coaching program for youth with chronic illness. It is a one-on-one intervention targeting skills in stress management, goal-setting, cognitive reframing, and meaning-making. We aimed to (i) assess the feasibility and acceptability of PRISM and (ii) explore PRISM's impact on clinical outcomes among youth with chronic musculoskeletal pain (CMP). This was a single-arm pilot trial of PRISM for youth with CMP aged 12-17 years. Patients completed patient-reported outcome measures (PROs) pre- and post- intervention; patients and caregivers provided qualitative feedback. Twenty-seven patients were enrolled (63% enrollment rate); 82% percent were female. The patients' median age was 16 years (IQR: 13-16). The intervention completion rate was 81% (n = 22). The mean satisfaction for PRISM overall was 4.3 (SD 0.9), while the mean acceptability of the intervention measure (AIM) was 4.4 (SD 0.89). Participants reported improved resilience (2.2 [SD 5.1]), functional disability (-3.5 [IQR: -6.0, 1.0]), and psychological distress (-1.0 [-5.0, 2.0]) from baseline to immediately post-treatment; pain intensity, pain catastrophizing, and global health were similar at both time points. Feedback was positive and suggested that a group component may be helpful. PRISM is feasible and acceptable among youth with CMP. Exploratory analyses suggest improvements in clinically relevant outcomes, warranting further investigation.

Recognizing medical child abuse in children presenting with chronic pain.

Medical child abuse (MCA), previously referred to as Munchausen by proxy, can present as chronic pain. We report the presentation of five children seeking treatment for chronic pain who we identified as victims of MCA. The index case had essentially not eaten for the 6 years of her life due to alleged allergies to all foods, developed severe pain, used a wheelchair for ambulation beyond a few blocks, and was alleged to have dysautonomia requiring oxygen monitoring at night. Other cases posed as arthritis that resulted in foot amputation and total body pain, fibromyalgia with alleged mutation negative Stickler syndrome who had symptoms only in her mother's presence, severe incapacitating intermittent pains along with abdominal pain that resulted in appendectomy, cholecystectomy, and pancreatectomy, and alleged disabling hypermobile Ehlers-Danlos in a non-hypermobile child for which the mother sought a power wheelchair. The unusual pattern to the pain, the presence of multiple additional, atypical symptoms and diagnoses, and a generally well appearing child are characteristic. The perpetrator is typically over-invested in the symptoms, derives tangible and intangible secondary gain from the child's alleged illnesses, and is able to present the child in such a fashion to enlist the physician to aid in perpetuating the abuse. These children are highly over-medicalized and suffer significant morbidity. Multiple barriers exist to identifying and reporting these children to Child Protective Services, which need to be recognized and overcome in order to protect these vulnerable children.

Characterizing Neurocognitive Impairment in Juvenile Fibromyalgia Syndrome: Subjective and Objective Measures of Dyscognition.

Objectives: Our understanding of brain fog, or dyscognition, among youth with juvenile fibromyalgia syndrome is limited. We aimed to determine the prevalence of subjective (self-reported) and objective dyscognition, as well as factors associated with subjective dyscognition in juvenile fibromyalgia syndrome. Methods: A cross-sectional cohort study of patients (n = 31) 12-17 years old diagnosed with primary juvenile fibromyalgia syndrome and one of their parents from 2017 to 2019. Subjects completed a series of survey measures and patients completed a brief neurocognitive battery. Subjective dyscognition was determined based on scores on the Pediatric Quality of Life Inventory (PedsQL) Cognitive Functioning Scale and Behavior Rating Inventory of Executive Function (BRIEF-2) global executive composite (GEC). Objective dyscognition was defined as impairment of more than two standard deviations in any of the neurocognitive domains. We used Fisher's exact test or Wilcoxon rank-sum test, as appropriate, to compare clinical patients based on the presence of dyscognition. Multivariable logistic regression modeling was performed to determine factors associated with subjective dyscognition. Results: Of the 31 subjects, 65% reported subjective dyscognition and 39% had objective dyscognition, primarily in the domains of psychomotor speed (23%), executive function (23%), and attention (3%). Subjective dyscognition was not indicative of objective dyscognition. Subjective dyscognition was independently associated with functional disability (OR: 1.19 [95% CI: 1.02-1.40]) and anxiety (OR: 1.12 [95% CI: 1.02-1.24]). Discussion: Adolescents with fibromyalgia predominantly experience subjective dyscognition but more than 1/3 also experience objective dyscognition. Future research should explore the impact of interdisciplinary rehabilitation programs on the treatment of dyscognition in youth with JFMS.

Suicidal risk and resilience in juvenile fibromyalgia syndrome: a cross-sectional cohort study.

BACKGROUND: To characterize suicidality among youth with juvenile fibromyalgia syndrome (JFMS) receiving treatment from pediatric rheumatologists at a tertiary care center in order to determine the prevalence of suicidality in JFMS and to explore risk factors for persistent suicidal ideation. METHODS: We performed a cross-sectional cohort study of children 12-17 years old with JFMS seen in a specialty pediatric rheumatology pain clinic from 7/2017-9/2019. All subjects completed patient-reported outcomes measures, complemented by retrospective chart review. Subjects who endorsed item 8 on the Children's Depression Inventory, 2nd Edition (CDI-2) were categorized as endorsing suicidal ideation. We assessed for differences between the suicidal and non-suicidal patients using Wilcoxon-rank sum test. Logistic regression modeling was performed to identify psychosocial factors associated with suicidality. RESULTS: Of the 31 subjects, more than one-quarter endorsed suicidality. Nearly 90% of teens with suicidal ideation were established in outpatient counseling. In bivariate analyses, suicidality was associated with lower resilience and greater depression and anxiety (all p < 0.05). Pain intensity trended towards a statistically significant positive association (OR: 1.16 [0.99-1.37]; p = 0.06). Lower resilience was independently associated with suicidality (OR: 0.90 [95% CI: 0.82-0.98]; p < 0.02). CONCLUSIONS: Suicidality was prevalent among youth with JFMS and persistent despite concurrent receipt of mental health services. Higher patient-level resilience was independently associated with a reduced odds of suicidality. Future work should examine the role of resilience training on reducing psychological distress and mitigating the risk of suicidality in JFMS.

Longitudinal Impact of Parental Catastrophizing on Child Functional Disability in Pediatric Amplified Pain.

OBJECTIVE: Children with amplified musculoskeletal pain (AMPS) experience significant functional disability, with impairment in their ability to participate in age-appropriate activities of daily living. Parental factors play an important role in a child's pain symptoms and treatment outcomes, with parental pain catastrophizing and protective behaviors linked to several maladaptive outcomes for children. Aims of the current study were to examine how parental pain catastrophizing, child pain catastrophizing, and parental protective behaviors longitudinally impacted functional disability for children with AMPS. METHODS: Archival data were examined from parent-child dyads presenting to a tertiary pain clinic for treatment of AMPS. Over 1 year, parents completed measures assessing the level of pain catastrophizing, common behavioral responses to child pain, and child functional disability. Children completed measures of pain catastrophizing and functional disability. Measures were collected at initial evaluation, 6-months, and 12-months. Latent growth models (LGM) were conducted to examine how to study variables longitudinally impacted the rate of change in child functional disability. RESULTS: Examining a comprehensive LGM of study variables, parental catastrophizing emerged as the sole contributing factor to slower improvement in functional disability. CONCLUSIONS: The strong influence of parental pain catastrophizing on functional disability may relate to parents limiting behaviors that promote adaptive coping in children with pain. As such, parents who catastrophize may benefit from specific interventions to increase their use of adaptive behavioral responses, such as redirecting children to complete functional activities and encouraging the use of positive coping skills for pain-related distress.

Development and Psychometric Evaluation of the PROMIS Pediatric Pain Intensity Measure in Children and Adolescents with Chronic Pain.

The goal of the current study was to enhance the measurement of the pediatric chronic pain experience through a methodologically rigorous approach. This paper outlines the development and initial validation of a pain intensity measure for pediatric patients with chronic pain using Patient-Reported Outcomes Measurement Information System methodology. Measure development incorporated feedback from children with painful conditions. Based on input from pediatric participants and content experts, 4 candidate items assessing pain intensity were included for large scale testing. Children completed self-report items pertaining to their pain experience that were developed as part of a larger pool of new candidate Patient-Reported Outcomes Measurement Information System pediatric pain domain items as well as measures of pain interference, depressive symptoms, fatigue, pain behavior, pain intensity, and pain catastrophizing. The final sample for the large scale testing included N = 442 pediatric patients between the ages 8 to 18 years (Mean age = 13.54, Standard Deviation = 2.78; 71.27% female) experiencing chronic pain. Psychometric analysis resulted in a final measure that included 3 items with evidence of reliability (Cronbach alpha = .82) and convergent validity. The Likert format of the response options may be preferable to the traditional numeric rating scale for use in pediatric populations who experience chronic pain based on patients' feedback, which was directly utilized in designing the scale. Further, the inclusion of fewer and clinically meaningful response options should reduce ambiguity for young respondents. PERSPECTIVE: We have developed and evaluated a clinically sensitive and psychometrically precise 3-item pain intensity measure with Likert-type responses for self-report use among children and adolescents ages 8 to 18 years with chronic pain. Development of the item content and response options included input from children and adolescents with chronic pain. The development of pain intensity items with pediatric appropriate language, and labeled, fewer response options to yield maximal clinically meaningful information improves the precision of pain intensity measurement in children.

The spectrum of pediatric amplified musculoskeletal pain syndrome.

BACKGROUND: Children presenting with musculoskeletal pain to pediatric rheumatology clinics are very heterogeneous and on a continuum from those with localized pain to total body pain. Many report intermittent, rather than constant, pain. We examined clinical and psychological characteristics of these children at presentation and specifically those who fulfilled the criteria for fibromyalgia. METHODS: We performed a retrospective, cross-sectional cohort study of children under ≤18 years old presenting to the pediatric rheumatology pain clinic between January 2015 and July 2019 and enrolled in a patient registry. We included children diagnosed with amplified pain, excluding those fulfilling criteria for complex regional pain syndrome. Abstracted data included clinical characteristics, pain symptoms, functional disability inventory (FDI), widespread pain index, and symptom severity scale. RESULTS: We analyzed 636 subjects, predominantly non-Hispanic Caucasian females. Using median split method, 54% had diffuse pain (≥ 5 body regions involved), but, of these, only 58% met criteria for fibromyalgia. Subjects with diffuse pain, compared to those with localized pain had a longer duration of pain (24 vs 12 months, p < 0.01), reported greater pain intensity (6/10 vs 5/10, p < 0.001), greater mental health burden, and poorer function (FDI 25 vs 19, p < 0.0001). Subjects with limited pain more often reported a history of trigger event (34% vs 24%, p < 0.01) but not autonomic changes (14% vs 14%, p = 0.94). The presence of adverse childhood experiences did not differ among those with limited versus diffuse pain except for parental divorce (16% vs 23%, p = 0.03). Intermittent pain was reported in 117 children (18%) and, compared to subjects with constant pain, they reported less pain (0/10 vs 6/10) and were more functional (FDI 13 vs 25) (both p < 0.0001). CONCLUSIONS: There exists a wide spectrum of pain manifestations among children with amplified pain including limited or diffuse and constant or intermittent pain. Most children who presented to our clinic did not fulfill criteria for fibromyalgia but nonetheless had significant symptoms and disability. Studies focusing on fibromyalgia may miss the full extent of childhood amplified pain. Additionally, research limited to those meeting the fibromyalgia criteria likely underestimate the significant impact of amplified pain among the pediatric population.

Erythema Ab Igne in an Adolescent With Chronic Pain: An Alarming Cutaneous Eruption From Heat Exposure.

Erythema ab igne (EAI) is a cutaneous finding caused by prolonged heat exposure and is characterized by a reticular, brownish-pigmented, often telangiectatic dermatosis. The eruption is reminiscent of livedo reticularis, which is typically seen in the setting of a number of rheumatologic conditions, most prominently vasculitis. Identification of key features distinguishing EAI from livedo reticularis can aid in the diagnosis of EAI and correct elucidation of the underlying etiology. Our patient presented with heating pad-induced EAI in the setting of chronic pain. Only 6 other pediatric cases of EAI associated with heat sources for chronic pain are reported (Acta Derm Venereol. 2014;94:365-367, J Pediatr. 2013;163:1789, Int J Eat Disord. 2013;46:381-383, Arch Dis Child. 2008;93:389, Arch Pediatr Adolesc Med. 2012;166:185-186, Br J Clin Pract. 1990;44:248-251). Our case highlights the need for awareness of this pathognomonic skin eruption in children with chronic pain conditions to help avoid an extensive workup for vasculitis.

Retrospective review of immobilization vs. immediate resumption of activity in patients with Oligoarticular juvenile idiopathic arthritis following knee injections.

BACKGROUND: Intraarticular corticosteroid injection (IACI) is one of the most common treatments in oligoarticular Juvenile Idiopathic Arthritis (JIA). Activity recommendations following injection vary, as there are no published studies on splinting JIA patients post-IACI (splinting is a form of rest). Texas Scottish Rite Hospital for Children (TSRH) splints patients post-IACI for 24 h while The Children's Hospital of Philadelphia (CHOP) does not. The aim of this study was to compare the number of cases of recurrent arthritis following IACI between these two post-injection practices. METHODS: Data were retrospectively collected at CHOP and TSRH. Patients diagnosed with oligoarticular JIA according to International League of Associations for Rheumatology (ILAR) criteria (2nd revision, 2001) between 2008 and 2010 were included. Bivariate analysis (Wilcoxon rank-sum tests, chi-squared tests) was run to assess differences in outcomes by site. Inverse probability of treatment weighted Cox regression was employed to adjust for site differences. RESULTS: The population at TSRH was younger than at CHOP (p < 0.05) and had more whites (p = 0.03). Disease duration was significantly longer at TSRH than at CHOP (0.40 vs. 0.74 years, p = 0.014). More children were on biologics at the time of injection at CHOP (p < 0.05). The baseline physician global (p < 0.001) was higher at CHOP, as was the joint disease severity (p < 0.001). CHOP had fewer reoccurrences of knee arthritis compared to TSRH: 26% vs 38% (p = 0.14). CONCLUSIONS: The baseline populations were different in that the TSRH group had more whites and Hispanics, were younger and, perhaps, had less severe disease than CHOP. Patients treated with post-injection splinting had a trend toward more arthritis reoccurrence (38% vs. 26%, p = 0.14). Splinting is not clearly beneficial post-injection. TRIAL REGISTRATION: This is an observational study, so it is not applicable.

Patient-proxy agreement on health-related quality of life in juvenile fibromyalgia syndrome.

BACKGROUND: Health-related quality of life (HRQoL) measures serve as important indicators of pain-related physical and psychosocial function in youth with juvenile fibromyalgia syndrome (JFMS). While the administration of parent-proxy reported HRQoL measures in the assessment of JFMS is common, its added clinical value to patient self-reports is unclear. We aimed to determine the level of agreement on HRQoL among patients with JFMS as well as their parent-proxies and to determine factors associated with this agreement. METHODS: We performed a retrospective, cross-sectional cohort study of children aged 8 to 17 years diagnosed with JFMS and presenting for initial evaluation to a pediatric rheumatology pain clinic between April 2017 and May 2018. All patients and proxies were administered the Pediatric Quality of Life Short Form 15 Generic Core Scales (PedsQL SF-15) as part of routine clinical care. We calculated absolute discrepancy scores (absolute value of parent-proxy score minus patient score) to describe the extent of difference in HRQoL scores between parent-proxies and patients. We examined agreement between parent-proxy report and patient self-report via intraclass correlation coefficients (ICCs), stratified by age and sex, as well as Bland-Altman plots. We also used multivariate regression models to determine factors associated with level of agreement. RESULTS: A total of 65 patient-proxy pairs were included in this study. ICCs demonstrated good to excellent agreement between all parent-proxy and patient measures of HRQoL irrespective of the patient's age or sex. The level of agreement was not associated with pain duration or pain severity but less agreement on psychosocial HRQoL was associated with older patient age (ß = 1.30; p < 0.05). CONCLUSIONS: This study in youth with JFMS demonstrated good to excellent patient-proxy agreement across all domains of the PedsQL SF-15 irrespective of patient's age or sex. Our findings suggest that parent-proxy reports do not provide additional information beyond that obtained from the patient self-report of HRQoL according to the PedsQL SF-15. In order to facilitate children and adolescents with JFMS becoming partners in their own healthcare, and to decrease the burden of multiple questionnaires, we propose focusing on patients' own perceptions of HRQoL in the clinical setting.

The Role of Patient and Parental Resilience in Adolescents with Chronic Musculoskeletal Pain.

OBJECTIVES: To assess the level of resilience among patients with chronic musculoskeletal pain and their parents and to determine factors associated with patient and parental resilience. STUDY DESIGN: Cross-sectional cohort study of children aged 13-17 years diagnosed with chronic musculoskeletal pain and their parents. Patient-parent pairs were seen for initial consultation in the pediatric rheumatology pain clinic at Children's Hospital of Philadelphia between March and May 2018 and were administered a series of questionnaires including measures of resilience (Connor-Davidson Resilience Scale 10 item, The 14-item Resilience Scale, and the 7Cs of Resilience Tool). We calculated Pearson correlation coefficients to examine the relationship between the variables of interest and resilience. RESULTS: According to all resilience measures, patients and parents had low to moderate levels of resilience. These levels were lower than those previously reported among healthy populations, as well as those with chronic medical conditions. According to the Connor-Davidson Resilience Scale 10 item, patient-level resilience was negatively correlated with pain level (r = -0.48), physical disability (r = -0.54), and symptom severity (r = -0.53). The level of resilience among patients was positively correlated with energy level (r = 0.57) and health-related quality of life (r = 0.64). Parental resilience was positively correlated with parental mental health (r = 0.61). CONCLUSIONS: Higher patient resilience was correlated with reduced disease severity among adolescents with chronic musculoskeletal pain. Future research should explore whether fostering resilience in adolescents with chronic musculoskeletal pain via the application of resilience-training interventions mitigates disease burden in this vulnerable patient population.

Intraarticular Glucocorticoid Injection as Second-line Treatment for Lyme Arthritis in Children.

OBJECTIVE: To determine whether second-line intraarticular glucocorticoid (IAGC) injection improves outcomes in children with persistently active Lyme arthritis after initial antibiotics. METHODS: We conducted an observational comparative effectiveness study through chart review within 3 pediatric rheumatology centers with distinct clinical approaches to second-line treatment of Lyme arthritis. We primarily compared children receiving second-line IAGC to children receiving a second course of antibiotics alone. We evaluated the risk of developing antibiotic-refractory Lyme arthritis (ARLA) using logistic regression and the time to clinical resolution of Lyme arthritis using Cox regression. RESULTS: Of 112 children with persistently active Lyme arthritis after first-line antibiotics, 18 children received second-line IAGC (13 with concomitant oral antibiotics). Compared to children receiving second-line oral antibiotics alone, children treated with IAGC had similar baseline characteristics but lower rates of ARLA (17% vs 44%; OR 0.3, 95% CI 0.1-0.95; p = 0.04) and faster rates of clinical resolution (HR 2.2, 95% CI 1.2-3.9; p = 0.01). Children in IAGC and oral antibiotic cohorts did not differ in treatment-associated adverse events. Among children receiving second-line IAGC, outcomes appeared similar irrespective of use of concomitant antibiotics. Outcomes were also similar between intravenous (IV) and oral antibiotic-treated cohorts, but older children seemed to respond more favorably to IV therapy. IV antibiotics were also associated with higher rates of toxicity. CONCLUSION: IAGC injection appears to be an effective and safe second-line strategy for persistent Lyme arthritis in children, associated with rapid clinical resolution and reduced need for additional treatment.

Pediatric Antibiotic-refractory Lyme Arthritis: A Multicenter Case-control Study.

OBJECTIVE: Few factors have consistently been linked to antibiotic-refractory Lyme arthritis (ARLA). We sought to identify clinical and treatment factors associated with pediatric ARLA. METHODS: We performed a case-control study in 3 pediatric rheumatology clinics in a Lyme-endemic region (2000-2013). Eligible children were aged ≤ 18 years with arthritis and had positive testing for Lyme disease by Western blot. Cases were 49 children with persistently active arthritis despite ≥ 8 weeks of oral antibiotics or ≥ 2 weeks of parenteral antibiotics; controls were 188 children whose arthritis resolved within 3 months of starting antibiotics. We compared preselected demographic, clinical, and treatment factors between groups using logistic regression. RESULTS: Characteristics positively associated with ARLA were age ≥ 10 years, prolonged arthritis at diagnosis, knee-only arthritis, and worsening after starting antibiotics. In contrast, children with fever, severe pain, or other signs of systemic inflammation were more likely to respond quickly to treatment. Secondarily, low-dose amoxicillin and treatment nonadherence were also linked to higher risk of ARLA. Greater antibiotic use for children with ARLA was accompanied by higher rates of treatment-associated adverse events (37% vs 15%) and resultant hospitalization (6% vs 1%). CONCLUSION: Older children and those with prolonged arthritis, arthritis limited to the knees, or poor initial response to antibiotics are more likely to have antibiotic-refractory disease and treatment-associated toxicity. Children with severe symptoms of systemic inflammation have more favorable outcomes. For children with persistently active Lyme arthritis after 2 antibiotic courses, pediatricians should consider starting antiinflammatory treatment and referring to a pediatric rheumatologist.

The pediatric rheumatology objective structured clinical examination: progressing from a homegrown effort toward a reliable and valid national formative assessment.

BACKGROUND: Of 37 pediatric rheumatology fellowship training programs in the United States, many have three or fewer fellows at a given time, making large-scale assessment of fellow performance difficult. An objective structured clinical examination (OSCE) is a scenario-based simulation method that assesses individual performance, thus indirectly measuring training program effectiveness. This study describes the development and implementation of two national pediatric rheumatology OSCEs and methods used for programmatic improvement. METHODS: OSCEs for pediatric rheumatology fellows were held in 2009 and 2011 during national rheumatology meetings using scenarios and assessment forms originally developed by a fellowship program director. The seven scenarios tested medical knowledge, physical exam and interpersonal skills. Pediatric rheumatologist evaluators assessed fellows' performance using checklists and gave immediate feedback. Program directors were sent summaries of their fellows' performances. Fellows evaluated the OSCE, providing organizational and scenario improvement suggestions. Programmatic changes to the 2011 OSCE were based on 2009 performance data and program evaluation feedback. RESULTS: Twenty-two fellows participated in 2009 and 19 in 2011. Performance scores in similar scenarios did not change considerably over the two iterations. In 2009, 85.7% of participants reported desire to change clinical behavior. Assessors' 2009 program evaluation data prompted changes in rating scales and removal of invalid or unreliable assessments. Negative evaluation data about individual stations decreased from 60% in 2009 to 15.4% in 2011. Fellows' ratings of the experience's overall value were similar in 2009 and 2011. The average experience ratings were lower among fellows who proposed scenario-specific improvements and higher among those who recommended organizational improvements. CONCLUSIONS: The 2011 examination exhibited programmatic improvement via reduction in fellows' scenario-specific negative feedback. Fellows' overall satisfaction did not change. Further work in scenario selection, assessment validation and inter-rater reliability will improve future pediatric rheumatology OSCEs.