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Cytosolic 5′-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis

Lookup NU author(s): Dr James Miller, Professor Michael Hanna

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This work is licensed under a Creative Commons Attribution 4.0 International License (CC BY 4.0).


Abstract

© 2017 BMJ Publishing Group Ltd & European League Against Rheumatism. Objectives Autoantibodies directed against cytosolic 50-nucleotidase 1A have been identified in many patients with inclusion body myositis. This retrospective study investigated the association between anticytosolic 50-nucleotidase 1A antibody status and clinical, serological and histopathological features to explore the utility of this antibody to identify inclusion body myositis subgroups and to predict prognosis. Materials and methods Data from various European inclusion body myositis registries were pooled. Anticytosolic 50-nucleotidase 1A status was determined by an established ELISA technique. Cases were stratified according to antibody status and comparisons made. Survival and mobility aid requirement analyses were performed using Kaplan-Meier curves and Cox proportional hazards regression. Results Data from 311 patients were available for analysis; 102 (33%) had anticytosolic 50-nucleotidase 1A antibodies. Antibody-positive patients had a higher adjusted mortality risk (HR 1.89, 95% CI 1.11 to 3.21, p=0.019), lower frequency of proximal upper limb weakness at disease onset (8% vs 23%, adjusted OR 0.29, 95% CI 0.12 to 0.68, p=0.005) and an increased prevalence of excess of cytochrome oxidase deficient fibres on muscle biopsy analysis (87% vs 72%, adjusted OR 2.80, 95% CI 1.17 to 6.66, p=0.020), compared with antibody-negative patients. Interpretation Differences were observed in clinical and histopathological features between anticytosolic 50-nucleotidase 1A antibody positive and negative patients with inclusion body myositis, and antibody-positive patients had a higher adjusted mortality risk. Stratification of inclusion body myositis by anticytosolic 50-nucleotidase 1A antibody status may be useful, potentially highlighting a distinct inclusion body myositis subtype with a more severe phenotype.


Publication metadata

Author(s): Lilleker JB, Rietveld A, Pye SR, Mariampillai K, Benveniste O, Peeters MTJ, Miller JAL, Hanna MG, Machado PM, Parton MJ, Gheorghe KR, Badrising UA, Lundberg IE, Sacconi S, Herbert MK, McHugh NJ, Lecky BRF, Brierley C, Hilton-Jones D, Lamb JA, Roberts ME, Cooper RG, Saris CGJ, Pruijn GJM, Chinoy H, van Engelen BGM

Publication type: Article

Publication status: Published

Journal: Annals of the Rheumatic Diseases

Year: 2017

Volume: 76

Issue: 5

Pages: 862-868-

Print publication date: 01/05/2017

Online publication date: 25/01/2017

Acceptance date: 05/11/2016

ISSN (print): 0003-4967

ISSN (electronic): 1468-2060

Publisher: BMJ Publishing Group

URL: https://doi.org/10.1136/annrheumdis-2016-210282

DOI: 10.1136/annrheumdis-2016-210282


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