A possible case of Churg-Strauss syndrome in a 9-year-old child

Liu, Jinling; Xu, Yingchun; Chen, Zhimin; Xu, Xuefeng; Lu, Meiping; Wang, Yingshuo; Zhou, Yunlian; Gu, Weizhong

doi:10.6061/clinics/2012(08)22

Cited by 5 publications

(2 citation statements)

References 13 publications

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“…Literature on pediatric EGPA is scarce, and the specificities of pediatric cases remain poorly described. To the best of our knowledge, only 38 articles describing pediatric cases of EGPA are listed in the PubMed database, reporting 92 cases (mostly individual case reports) . The two largest reported pediatric EGPA series described, respectively, 8 and 13 pediatric cases.…”

Section: Introductionmentioning

confidence: 99%

Eosinophilic granulomatosis with polyangiitis in children: Data from the French RespiRare® cohort

Fina

Dubus

Tran

et al. 2018

Pediatric Pulmonology

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This French cohort is the biggest pediatric EGPA series described to date, with a long follow-up period. The findings confirm that pediatric EGPA has specific clinical, radiological, and histological characteristics that differ from adult EGPA. Development of systemic symptoms, and consequently diagnosis, occur with a shorter delay in children, mainly during the eosinophilic phase and leading to a specific presentation.

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Section: Introductionmentioning

confidence: 99%

Eosinophilic granulomatosis with polyangiitis in children: Data from the French RespiRare® cohort

Fina

Dubus

Tran

et al. 2018

Pediatric Pulmonology

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“…An examination of large contemporary databases reveals childhood-onset EGPA (cEGPA) accounts for less than 2% of all cases of pediatric vasculitis (CARRANet, ARChiVe unpublished data). Only 38 cases of cEGPA have been published, with most reports detailing experience with a single patient 9,10,11,12,13,14 . It is suspected that children with EGPA are less likely than adult patients to have positive ANCA titers, more likely to have pulmonary involvement, and suffer higher mortality rates 9 .…”

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confidence: 99%

Childhood-onset Eosinophilic Granulomatosis with Polyangiitis (formerly Churg-Strauss Syndrome): A Contemporary Single-center Cohort

Gendelman

Zeft²,

Spalding³

2013

J Rheumatol

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Cutaneous manifestations of childhood Eosinophilic Granulomatosis with Polyangiitis (cEGPA): A case‐based review

Bridges

Shenk

Martin

et al. 2020

Pediatric Dermatology

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Background/ObjectivesThis study seeks to better define the clinical presentation and histopathology of cutaneous manifestations in childhood eosinophilic granulomatosis with polyangiitis (cEGPA).MethodsCase reports were collected from Ovid Medline Database and PubMed using keyword identifiers from 1946 to 2017. Adult patients ≥ 18 years and cases not diagnosed with EGPA by the author were excluded. Sixty‐five case reports of cEGPA were initially identified. These were reviewed individually, and fifty cases were determined to meet the American College of Rheumatology criteria for EGPA. No case series examining the cutaneous morphology and histopathology were identified. Cutaneous morphology, lesion location, and cutaneous histopathology results were recorded. Results were analyzed using summary statistics.ResultsSixty‐four percent (32/50) of cEGPA patients presented with cutaneous manifestations. Twenty‐nine cases provided specific morphological descriptions and lesion location. Common manifestations included purpura (15/29), subcutaneous nodules (8/29), and a macular/papular/maculopapular rash (8/29). However, twelve different cutaneous morphologies were identified in this review. Lesions occurred most commonly on the extremities (26/29). Twenty‐two cases reported corresponding cutaneous histopathology, which revealed extravascular eosinophils (15/22), vasculitis (13/22), and granulomas (5/22). Only one biopsy sample (1/22) had all three classical EGPA characteristics of granulomas, extravascular eosinophils, and vasculitis.ConclusionWith nearly two‐thirds of cEGPA patients presenting with cutaneous manifestation, this study highlights the importance of clinical recognition of this disease by dermatologists. While the varied morphology of skin lesions and rarity of this disease makes cEGPA a difficult diagnosis, prompt recognition and treatment will improve outcomes in this patient population.

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A possible case of Churg-Strauss syndrome in a 9-year-old child

Cited by 5 publications

References 13 publications

Eosinophilic granulomatosis with polyangiitis in children: Data from the French RespiRare® cohort

Eosinophilic granulomatosis with polyangiitis in children: Data from the French RespiRare® cohort

Childhood-onset Eosinophilic Granulomatosis with Polyangiitis (formerly Churg-Strauss Syndrome): A Contemporary Single-center Cohort

Cutaneous manifestations of childhood Eosinophilic Granulomatosis with Polyangiitis (cEGPA): A case‐based review

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