First name
Sabrina
Last name
Gmuca

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

Exploring the intersection of adverse childhood experiences, pediatric chronic pain, and rheumatic disease.

Year of Publication

2022

Number of Pages

14

Date Published

2022 Feb 14

ISSN Number

1546-0096

Abstract

<p><strong>BACKGROUND: </strong>While the general relationship between ACEs and the development of chronic pain has become increasingly clear, how ACEs may shape a child's clinical presentation with regards to chronic pain has yet to be fully expounded. We aimed to determine the association between ACEs and clinical manifestations of pediatric chronic pain and explore the interaction of ACEs and pediatric rheumatic disease among youth with chronic pain on health-related outcomes.</p>

<p><strong>METHODS: </strong>We conducted a cross-sectional cohort study of patients aged ≤18 years with chronic pain seen in a pediatric rheumatology amplified pain clinic between August 2018 and July 2020. We stratified subjects into three groups: no ACEs, one ACE, and ≥ 2 ACEs. We assessed clinical signs and symptoms associated with the presence of ACEs using Chi-square or Wilcoxon-rank test. The association between ACEs as well as other variables of interest with functional impairment was tested using simple and multivariable linear regression.</p>

<p><strong>RESULTS: </strong>Of the 412 patients included, more than 75% of patients reported at least one ACE. Most frequent included history of mental illness in a first degree relative (56%) and parental divorce or separation (20%). Those with ≥2 ACEs had more somatic symptoms, worse functional disability, and a higher proportion of mental health conditions. There appeared to be a dose dependent interaction between ACEs and functional disability from co-morbid rheumatologic disease. In multivariable regression, higher verbal pain score, symptom severity score (SSS), and presence of autonomic changes were associated with estimated average increase in FDI score (β = 1.05, 1.95 and 4.76 respectively; all p&nbsp;&lt; 0.01).</p>

<p><strong>CONCLUSION: </strong>Children with chronic pain and/or rheumatologic diseases who are exposed to ACEs are at increased risk of greater symptomatology, functional disability, and somatization of symptoms. Our findings indicate an ongoing need for systemic evaluation of ACEs in children with chronic pain and/or rheumatic disease and incorporation of trauma-based care.</p>

DOI

10.1186/s12969-022-00674-x

Alternate Title

Pediatr Rheumatol Online J

PMID

35164793

Title

Evaluation of pediatric rheumatology telehealth satisfaction during the COVID-19 pandemic.

Year of Publication

2021

Number of Pages

170

Date Published

2021 Dec 09

ISSN Number

1546-0096

Abstract

<p><strong>BACKGROUND: </strong>During the Coronavirus disease 2019 pandemic, ambulatory pediatric rheumatology healthcare rapidly transformed to a mainly telehealth model. However, pediatric patient and caregiver satisfaction with broadly deployed telehealth programs remains largely unknown. This study aimed to evaluate patient/caregiver satisfaction with telehealth and identify the factors associated with satisfaction in a generalizable sample of pediatric rheumatology patients.</p>

<p><strong>METHODS: </strong>Patients with an initial telehealth video visit with a rheumatology provider between April and June 2020 were eligible. All patients/caregivers were sent a post-visit survey to assess a modified version of the Telehealth Usability Questionnaire (TUQ) and demographic and clinical characteristics. TUQ total and sub-scale (usefulness, ease of use, effectiveness, satisfaction) scores were calculated and classified as "positive" based on responses of "agree" or "strongly agree" on a 5-point Likert scale. Results were analyzed using standard descriptive statistics and Wilcoxon signed rank testing. The association between demographic and clinical characteristics with TUQ scores was assessed using univariate linear regression.</p>

<p><strong>RESULTS: </strong>597 patients/caregivers met inclusion criteria, and the survey response rate was 42% (n = 248). Juvenile idiopathic arthritis was the most common diagnosis (33.5%). The majority of patients were diagnosed greater than 6 months previously (72.6%) and were prescribed chronic medications (59.7%). The median total TUQ score was 4 (IQR: 4-5) with positive responses in 81% of items. Of the subscales, usefulness scores were lowest (median: 4, p &lt; 0.001). Telehealth saves time traveling was the highest median item score (median = 5, IQR: 4-5). Within subscales, items that scored significantly lower included convenience, providing for needs, seeing rheumatologist as well as in person, and being an acceptable way to receive rheumatology services (all p &lt; 0.001). There were no significant demographic or clinical features associated with TUQ scores.</p>

<p><strong>CONCLUSIONS: </strong>Our results suggest telehealth is a promising mode of healthcare delivery for pediatric rheumatic diseases but also identifies opportunities for improvement. Innovation and research are needed to design a telehealth system that delivers high quality and safe care that improves healthcare outcomes. Since telehealth is a rapidly emerging form of pediatric rheumatology care, improved engagement and training of patients, caregivers, and providers may help improve the patient experience in the future.</p>

DOI

10.1186/s12969-021-00649-4

Alternate Title

Pediatr Rheumatol Online J

PMID

34886863

Title

Validation of Coding Algorithms for the Identification of Herpes Zoster among Children.

Year of Publication

2021

Date Published

2021 May 07

ISSN Number

1099-1557

Abstract

<p><strong>PURPOSE: </strong>To examine the validity of International Classification of Diseases, 10th Revision, (ICD-10) code-based algorithms for herpes zoster (HZ) in the electronic medical record (EMR) of a large, integrated pediatric healthcare network and to examine baseline demographics and chronic comorbidities associated with HZ in a representative pediatric population.</p>

<p><strong>METHODS: </strong>We reviewed the electronic charts of all patients with a single ICD-10 for HZ (B02.xx) as their primary or secondary diagnosis in the EMR of the Children's Hospital of Philadelphia (CHOP) healthcare network from January 2010 - March 2019. The positive predictive value (PPV) for a single code for HZ was calculated and alternative algorithms were examined to determine which method resulted in the highest PPV.</p>

<p><strong>RESULTS: </strong>The PPV for a single ICD-10 code was 91.7% (95% CI 80.8-95.4) for definitive and/or probable cases of HZ and 63.9% (95% CI 53.4-75.5%) for definitive cases alone. Adding a prescription for an antiviral did not improve the PPV. However, adding a new code for rash entered within 1 week of the HZ code increased the PPV to 100% for definitive and/or probable cases but with substantial loss of sensitivity. A high proportion of children with HZ who required inpatient hospitalization had chronic disease (70%) and were on systemic immunomodulatory therapy (50%).</p>

<p><strong>CONCLUSIONS: </strong>HZ can be identified with a high PPV in electronic medical records of children using ICD-10 code alone. These findings lay the foundation for future pharmacoepidemiologic research to better understand risk factors for HZ infection.</p>

DOI

10.1002/pds.5264

Alternate Title

Pharmacoepidemiol Drug Saf

PMID

33960541

Title

From Theory to Patient Care: a model for the development, adaptation, and testing of psychosocial interventions for patients with serious illness.

Year of Publication

2021

Date Published

2021 Mar 05

ISSN Number

1873-6513

Abstract

<p>Psychosocial and supportive care interventions are a cornerstone of palliative care science, yet there is little published guidance regarding how to develop, test, adapt, and ultimately disseminate evidence-based interventions. Our objective was to describe the application of a single intervention-development model in multiple populations of patients with serious illness. Specifically, we use the "Promoting Resilience in Stress Management" (PRISM) intervention as an exemplar for how the Obesity Related Behavioral Intervention Trials (ORBIT) intervention-development model may be applied to: (1) create an initial palliative care intervention; (2) adapt an existing intervention for a new patient-population; (3) expand an existing intervention to include new content; and, (4) consider dissemination and implementation of a research-proven intervention. We began by identifying key psychological and social science theories and translating them a testable clinical hypothesis. Next, we conducted observational studies and randomized trials to design, refine, and standardize PRISM within unique patient-populations. We moved backwards in the ORBIT model when necessary to adapt or expand PRISM content and delivery-strategies to meet patient-reported needs. Finally, we began to explore PRISM's effectiveness using Dissemination and Implementation research methods. Key lessons include the need to ground intervention-development in evidence-based theory; involve patient, clinician, and other stakeholders at every phase of development; "meet patients where they are at" with flexible delivery strategies; invest in the time to find the right scientific premise and the right intervention content; and, perhaps most importantly, involve an interdisciplinary research team.</p>

DOI

10.1016/j.jpainsymman.2021.02.036

Alternate Title

J Pain Symptom Manage

PMID

33677072

Title

Disordered eating among adolescents with chronic pain: the experience of a pediatric rheumatology subspecialty pain clinic.

Year of Publication

2021

Number of Pages

16

Date Published

2021 Feb 16

ISSN Number

1546-0096

Abstract

<p><strong>BACKGROUND: </strong>Disordered eating and chronic pain often co-occur in adolescents, but the relationship between these conditions is not well understood. We aimed to determine the prevalence of and to identify the clinical characteristics associated with the presence of disordered eating among adolescents with chronic musculoskeletal pain (CMP) presenting to a pediatric rheumatology subspecialty pain clinic.</p>

<p><strong>METHODS: </strong>This was a retrospective cohort study of pediatric patients presenting to a pediatric rheumatology subspecialty pain clinic for an initial consultation from March 2018 to March 2019. We complemented data from an existing patient registry with secondary chart review for patients identified with disordered eating. We compared patient characteristics based on the presence or absence of disordered eating among adolescents with CMP. Logistic regression modeling was used to determine factors associated with disordered eating.</p>

<p><strong>RESULTS: </strong>Of the 228 patients who were seen for an initial consultation in the pain clinic in 1 year, 51 (22.4%) had disordered eating. Only eight (15.7%) of the 51 patients identified with disordered eating had a previously documented formal eating disorder diagnosis. Through multivariate logistic regression modeling, we found that disordered eating was associated with older age, higher functional disability, presence of abdominal pain, presence of gastrointestinal comorbidities, and presence of anxiety (all p&nbsp;&lt; 0.05).</p>

<p><strong>CONCLUSIONS: </strong>Adolescents with chronic pain, especially those who experience gastrointestinal issues, anxiety, and greater functional disability, should be evaluated for disordered eating by the treating clinician in order to ensure timely and appropriate treatment.</p>

DOI

10.1186/s12969-021-00506-4

Alternate Title

Pediatr Rheumatol Online J

PMID

33593387

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

Validation of claims-based diagnoses of adult and pediatric neuromyelitis optica spectrum disorder and variations in diagnostic evaluation and treatment initiation.

Year of Publication

2019

Number of Pages

101488

Date Published

2019 Nov 01

ISSN Number

2211-0356

Abstract

<p><strong>BACKGROUND: </strong>Neuromyelitis optica spectrum disorder (NMOSD) is a rare demyelinating disease in need of more studies to determine effective treatment regimens. The rarity of the disorder, however, makes large randomized-controlled trials challenging. Validation of the International Classification of Diseases, 9th Revision, Clinical Modification (ICD-9-CM) code for NMO could facilitate the use of large healthcare claims data for future research. We aimed 1) to determine the positive predictive value (PPV) of the ICD-9-CM code for NMO as well as evaluate case-finding algorithms for the identification of patients with NMO/NMOSD and 2) to compare the evaluation of and treatment for pediatric versus adult patients.</p>

<p><strong>METHODS: </strong>This was a multicenter retrospective cohort study of patients with ≥ 1 ICD-9 code for NMO seen at 3 pediatric and 2 adult United States medical centers from 2001-2016. Using a standardized data entry form, pediatric and adult neurologists and rheumatologists reviewed patients' medical records to determine whether patients fulfilled the 2006 criteria for NMO and/or the 2015 criteria for NMOSD in order to determine the positive predictive value (PPV) for the ICD-9-CM code. Demographic and clinical information was abstracted from patient medical records to ascertain variables then evaluated in case-based finding algorithms for further identification of patients with true NMO/NMOSD. We also evaluated differences in clinical characteristics between pediatric and adult patients using chi-squared or Fisher's exact tests, as appropriate, to assess for treatment variation.</p>

<p><strong>RESULTS: </strong>A single code for NMO had a PPV of 47% across all sites, with significant site variation (0-77%). The best case-finding algorithm included at least 5 codes as well as a documented hospitalization (PPV&nbsp;=&nbsp;=90% for children and PPV&nbsp;=&nbsp;92% for adults). Children were more likely to be evaluated by a rheumatologist or ophthalmologist, undergo magnetic resonance imaging of the orbits, and receive immunosuppressive and biologic agents than their adult counterparts. Rituximab was administered similarly among the two groups.</p>

<p><strong>CONCLUSION: </strong>The ICD-9 code for neuromyelitis optica (NMO) is inaccurate for identification of NMO/NMOSD. Using case-finding algorithms increases the PPV. The initial diagnostic evaluation and treatment of NMOSD differs significantly between children and adults.</p>

DOI

10.1016/j.msard.2019.101488

Alternate Title

Mult Scler Relat Disord

PMID

31706167

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