Evidence map›Paper›PMID 30378704›Full record

ArticleNeuropathology and applied neurobiology2019

Histological heterogeneity in a large clinical cohort of juvenile idiopathic inflammatory myopathy: analysis by myositis autoantibody and pathological features.

S A Yasin, P W Schutz, C T Deakin, E Sag, H Varsani, S Simou, L R Marshall, S L Tansley, N J McHugh, J L Holton and 3 more

Open access · hybridAbstract read
In one paragraph

Article in Neuropathology and applied neurobiology, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers.

0numbers the graph read from it
0cells of the map it votes in
28citing papers in PubMed
3.7field-weighted citation impact, top 6% of its field
1 · What the graph read from it

What it found

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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

28 citing papers in PubMed, 52 citations in OpenAlex.

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  20. Juvenile Dermatomyositis: New Clues to Diagnosis and Therapy.Current treatment options in rheumatology · 2021
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

13 authors at 5 institutions in 1 country.

S A YasinInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.
P W SchutzInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.ORCID 0000-0001-7962-1560
C T DeakinInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.
E SagInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.
H VarsaniInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.
S SimouInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.
L R MarshallInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.
S L TansleyDepartment of Pharmacy and Pharmacology, University of Bath, Bath, UK.
N J McHughDepartment of Pharmacy and Pharmacology, University of Bath, Bath, UK.
J L HoltonDepartment of Molecular Neuroscience, MRC Centre for Neuromuscular Diseases, UCL Institute of Neurology, London, UK.
L R WedderburnInfection, Immunity, Inflammation Programme, UCL GOS Institute of Child Health, London, UK.
T S JacquesDevelopmental Biology and Cancer Programme, Developmental Biology of Birth Defects, UCL GOS Institute of Child Health, London, UK.ORCID 0000-0002-7833-2158
UK Juvenile Dermatomyositis Research Group (UK and Ireland)
University College London · GBUniversity of Bath · GBGreat Ormond Street Hospital · GBGreat Ormond Street Hospital for Children NHS Foundation Trust · GBMRC Prion Unit · GB

Funding

Versus Arthritis 21552Wellcome Trust 085860
6 · The paper itself

Abstract

aimJuvenile idiopathic inflammatory myopathies have been recently reclassified into clinico-serological subgroups. Myopathological correlates of the subgroups are incompletely understood.

methodsWe studied muscle biopsies from 101 children with clinically and serologically defined juvenile idiopathic inflammatory myopathies from the UK JDM Cohort and Biomarker Study by applying the international JDM score tool, myopathological review and C5b-9 complement analysis.

resultsAutoantibody data were available for 90/101 cases with 18/90 cases positive for anti-TIF1γ, 15/90 anti-NXP2, 11/90 anti-MDA5, 5/90 anti-Mi2 and 6/90 anti-PmScl. JDM biopsy severity scores were consistently low in the anti-MDA5 group, high in the anti-Mi2 group, and widely distributed in the other groups. Biopsies were classified histologically as perifascicular atrophy (22/101), macrophage-rich necrosis (6/101), scattered necrosis (2/101), clustered necrosis (2/101), inflammatory fibre invasion (2/101), chronic myopathic change (1/101), diffuse endomysial macrophage infiltrates (40/101) and minimal change (24/101). MDA5 cases segregated with the minimal change group and showed no capillary C5b-9-deposition. The Mi2 group displayed high severity scores and a tendency towards sarcolemmal complement deposition. NXP2 and TIF1γ groups showed a variety of pathologies with a high proportion of diffuse endomysial macrophage infiltrates and a high proportion of capillary C5b-9 deposition.

conclusionWe have shown that juvenile idiopathic inflammatory myopathies have a spectrum of histopathological phenotypes and show distinct complement attack complex deposition patterns. Both correlate in some cases with the serological subtypes. Most cases do not show typical histological features associated with dermatomyositis (e.g. perifascicular atrophy). In contrast, more than half show relatively mild histopathological changes.

Indexed as

AutoantibodiesAutoantigensChildChild, PreschoolCohort StudiesFemaleHumansMaleMyositisPhenotypeAutoantibodiesAutoantigensanti-SRPdermatomyositisimmune mediated necrotizing myopathyJDM score toolpolymyositisprincipal component analysis

Identifiers

PMID30378704
PMCPMC6767402
OpenAlexW2899336052

What OpenQuestion holds

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LicenceCC BY
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.