First name
David
Middle name
D
Last name
Sherry

Title

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

Year of Publication

2022

Number of Pages

848009

Date Published

2022

ISSN Number

2296-2360

Abstract

<p><strong>Objectives: </strong>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.</p>

<p><strong>Methods: </strong>A cross-sectional cohort study of patients ( = 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.</p>

<p><strong>Results: </strong>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]).</p>

<p><strong>Discussion: </strong>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.</p>

DOI

10.3389/fped.2022.848009

Alternate Title

Front Pediatr

PMID

35281242

Title

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

Year of Publication

2021

Number of Pages

3

Date Published

2021 Jan 06

ISSN Number

1546-0096

Abstract

<p><strong>BACKGROUND: </strong>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.</p>

<p><strong>METHODS: </strong>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.</p>

<p><strong>RESULTS: </strong>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 &lt; 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 &lt; 0.02).</p>

<p><strong>CONCLUSIONS: </strong>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.</p>

DOI

10.1186/s12969-020-00487-w

Alternate Title

Pediatr Rheumatol Online J

PMID

33407630

Title

The spectrum of pediatric amplified musculoskeletal pain syndrome.

Year of Publication

2020

Number of Pages

77

Date Published

2020 Oct 12

ISSN Number

1546-0096

Abstract

<p><strong>BACKGROUND: </strong>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.</p>

<p><strong>METHODS: </strong>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.</p>

<p><strong>RESULTS: </strong>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 &lt; 0.01), reported greater pain intensity (6/10 vs 5/10, p &lt; 0.001), greater mental health burden, and poorer function (FDI 25 vs 19, p &lt; 0.0001). Subjects with limited pain more often reported a history of trigger event (34% vs 24%, p &lt; 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 &lt; 0.0001).</p>

<p><strong>CONCLUSIONS: </strong>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.</p>

DOI

10.1186/s12969-020-00473-2

Alternate Title

Pediatr Rheumatol Online J

PMID

33046102

Title

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

Year of Publication

2019

Number of Pages

21

Date Published

2019 May 09

ISSN Number

1546-0096

Abstract

<p><strong>BACKGROUND: </strong>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.</p>

<p><strong>METHODS: </strong>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.</p>

<p><strong>RESULTS: </strong>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 (β = 1.30; p &lt; 0.05).</p>

<p><strong>CONCLUSIONS: </strong>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.</p>

DOI

10.1186/s12969-019-0320-y

Alternate Title

Pediatr Rheumatol Online J

PMID

31072338

Title

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

Year of Publication

2019

Date Published

2019 Apr 10

ISSN Number

1097-6833

Abstract

<p><strong>OBJECTIVES: </strong>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.</p>

<p><strong>STUDY DESIGN: </strong>Cross-sectional cohort study of children aged 13-17&nbsp;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.</p>

<p><strong>RESULTS: </strong>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&nbsp;=&nbsp;-0.48), physical disability (r&nbsp;=&nbsp;-0.54), and symptom severity (r&nbsp;=&nbsp;-0.53). The level of resilience among patients was positively correlated with energy level (r&nbsp;=&nbsp;0.57) and health-related quality of life (r&nbsp;=&nbsp;0.64). Parental resilience was positively correlated with parental mental health (r&nbsp;=&nbsp;0.61).</p>

<p><strong>CONCLUSIONS: </strong>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.</p>

DOI

10.1016/j.jpeds.2019.03.006

Alternate Title

J. Pediatr.

PMID

30981421

Title

Opioid Prescribing and Polypharmacy in Children with Chronic Musculoskeletal Pain.

Year of Publication

2018

Date Published

2018 Jun 13

ISSN Number

1526-4637

Abstract

<p><strong>Objective: </strong>Thirty percent of adults with fibromyalgia receive an opioid, but the prevalence of opioid prescribing in pediatric chronic musculoskeletal pain is unknown. The aims of this study were to determine the prevalence of and factors associated with opioid exposure and polypharmacy among children with chronic musculoskeletal pain.</p>

<p><strong>Methods: </strong>In this retrospective cohort study using health care claims data from 2000 to 2013, the index date was the first ICD-9 code 729.1. Included subjects were ≥ 2 and &lt; 18 years old at the index date with two or more codes within 12 months and 18 months of continuous enrollment. Subjects with burns, sickle cell disease, or malignancy were excluded. Opioid exposure was defined as one or more prescriptions within six months before or any time after the index date. Polypharmacy was considered minor (2-4 medications) or major (≥5 medications).</p>

<p><strong>Results: </strong>Of 25,321 included subjects, 20% received an opioid and 26% experienced minor polypharmacy. Opioid exposure was associated with female sex (odds ratio [OR] = 1.27, P &lt; 0.01), Caucasian race (OR = 1.27, P &lt; 0.01), hospitalization (OR = 1.20, P &lt; 0.01), and visit with anesthesiology (OR = 1.97, P &lt; 0.01) or orthopedics (OR = 1.09, P &lt; 0.05). Mental health codes were associated with decreased odds of opioid exposure (all P &lt; 0.05). Children seen by a chiropractor or physiatrist had a reduced odds of receipt of an opioid (OR = 0.42 and 0.84, respectively, both P &lt; 0.01).</p>

<p><strong>Conclusions: </strong>Twenty percent of children with chronic musculoskeletal pain received an opioid. Twenty-six percent experienced polypharmacy, with the majority receiving 2-4 medications. Increased availability of psychological and nonpharmacologic services are potential strategies to reduce opioid exposure.</p>

DOI

10.1093/pm/pny116

Alternate Title

Pain Med

PMID

29905842

Title

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

Year of Publication

2018

Date Published

2018 Mar 12

ISSN Number

1535-1815

Abstract

<p>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.</p>

DOI

10.1097/PEC.0000000000001460

Alternate Title

Pediatr Emerg Care

PMID

29538269

Title

The Treatment of Juvenile Fibromyalgia with an Intensive Physical and Psychosocial Program.

Year of Publication

2015

Number of Pages

731-7

Date Published

2015 Sep

ISSN Number

1097-6833

Abstract

<p><strong>OBJECTIVE: </strong>To assess the short-term and 1-year outcomes of children with fibromyalgia treated with intensive physical and occupational therapy (PT/OT) and psychotherapy.</p>

<p><strong>STUDY DESIGN: </strong>Children with fibromyalgia seen at a tertiary care hospital were treated with 5-6 hours of intensive PT/OT daily and at least 4 hours of psychosocial services weekly. All medications used for fibromyalgia were discontinued. Children underwent standardized testing, including a visual analog scale for pain; the Bruininks-Oseretsky Test of Motor Performance, Second Edition; the Bruce treadmill protocol; the Functional Disability Inventory; the Pain Stages of Change Questionnaire, adolescent version; and the Pediatric Quality of Life Inventory, Teen Report, at 3 time points: at program entry, at the end of the intensive program, and 1 year after the end of the program.</p>

<p><strong>RESULTS: </strong>Sixty-four children (median age, 16 years; 95% Caucasian; 94% female; median duration of symptoms, 21 months) were studied. The mean pain score decreased significantly from program entry to the end of the program (from 66 of 100 to 25 of 100; P = .001). At the 1-year follow-up, 33% reported no pain. All measures of function on the Bruininks-Oseretsky Test of Motor Performance, Second Edition improved significantly and remained at that level or continued to improve over the subsequent year. The mean Bruce treadmill protocol time first increased from 588 seconds to 801 seconds (P &lt; .001) and then dropped to 750 seconds (P = .005), which is at the 90th percentile for age and sex. All Pain Stages of Change Questionnaire, adolescent version subset scores improved significantly initially and were stable or improved at 1 year, as did the Pediatric Quality of Life Inventory, Teen Report total score.</p>

<p><strong>CONCLUSION: </strong>Children with fibromyalgia can be successfully treated without medications with a very intensive PT/OT and psychotherapy program. They have significantly improved pain and function by subject report and objective measures of function.</p>

DOI

10.1016/j.jpeds.2015.06.036

Alternate Title

J. Pediatr.

PMID

26209526

Title

Enthesitis in an inception cohort of enthesitis-related arthritis.

Year of Publication

2011

Number of Pages

1307-12

Date Published

2011 Sep

ISSN Number

2151-4658

Abstract

<p><strong>OBJECTIVE: </strong>To describe an enthesitis-related arthritis (ERA) inception cohort and determine which entheses and joints are most commonly affected.</p>

<p><strong>METHODS: </strong>We reviewed a retrospective inception cohort study of children with ERA who were diagnosed and treated at The Children's Hospital of Philadelphia between November 2007 and December 2009.</p>

<p><strong>RESULTS: </strong>During the study period, there were 32 newly diagnosed ERA patients. Fifty-nine percent were male, and the median age at the date of initial evaluation was 12.5 years (interquartile range [IQR] 10.2-14.3 years). The median number of tender entheses at presentation was 2 (IQR 0-5), and 21 subjects (66%) had at least 1 tender enthesis. The most prevalent tender entheses were the patellar ligament insertion at the inferior pole of the patella, the plantar fascial insertion at the calcaneus, the Achilles tendon insertion at the calcaneus, and the plantar fascial insertion at the metatarsal heads. Enthesitis was most often symmetric. The median number of active joints was 2 (IQR 0-4). The most commonly affected joints were the sacroiliacs, knees, and ankles. Sacroiliitis, which was defined clinically, was most often symmetric, while peripheral arthritis was most frequently asymmetric. The odds of having active enthesitis at 6 months increased significantly with each additional tender enthesis at the initial evaluation.</p>

<p><strong>CONCLUSION: </strong>Among pediatric patients with ERA, lower extremity enthesitis is prevalent at the time of diagnosis and is likely to persist 6 months later. Future studies should address standardization of the enthesitis examination, the pattern of enthesitis over time, enthesitis response to therapy, and the impact of enthesitis on quality of life.</p>

DOI

10.1002/acr.20508

Alternate Title

Arthritis Care Res (Hoboken)

PMID

21618453

Title

Enthesitis is an extraintestinal manifestation of pediatric inflammatory bowel disease.

Year of Publication

2012

Date Published

2012 Jan

ISSN Number

2146-2909

Abstract

<p><strong>BACKGROUND: </strong>Enthesitis is an extra-intestinal manifestation of inflammatory bowel disease (IBD) in adults. However, little has been published about the prevalence or characteristics of enthesitis in pediatric IBD.</p>

<p><strong>METHODS: </strong>We conducted a cross-sectional study of children and young adults ages 4-21 years with IBD. Subjects were recruited among those receiving routine care in a gastroenterology clinic. All subjects underwent a clinical examination of the entheses and joints, and completed a study questionnaire.</p>

<p><strong>RESULTS: </strong>We enrolled 43 subjects, who had a median age of 16 years and a median time from IBD diagnosis of 2.7 years. 32 subjects (74%) had Crohn disease, 10 subjects (23%) had indeterminate colitis, and 1 subject (2%) had ulcerative colitis. At least one tender enthesis was present in 21% of subjects and 12% had more than 2 tender entheses. The most commonly affected entheses were located at the inferior patella, the femoral greater trochanter, and the proximal humerus. The presence of enthesitis was associated with a higher intensity of recent musculoskeletal pain (p=0.03).</p>

<p><strong>CONCLUSIONS: </strong>Enthesitis is a prevalent extra-intestinal manifestation of pediatric IBD and is associated with increased musculoskeletal pain. Future studies should evaluate the functional and long-term impact of enthesitis on children with IBD.</p>

DOI

10.5455/apr.102920121510

Alternate Title

Ann Paediatr Rheumatol

PMID

24349861

WATCH THIS PAGE

Subscription is not available for this page.