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

Lilleker, JB, Rietveld, A, Pye, SR, Mariampillai, K, Benveniste, O, Peeters, MTJ, Miller, JAL, Hanna, MG, Machado, PM, Parton, MJ
et al (show 17 more authors) (2017) Cytosolic 5 '-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis. Annals of the Rheumatic Diseases, 76 (5). pp. 862-868.

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Objectives Autoantibodies directed against cytosolic 5′-nucleotidase 1A have been identified in many patients with inclusion body myositis. This retrospective study investigated the association between anticytosolic 5′-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 5′-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 5′-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 5′-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 5′-nucleotidase 1A antibody status may be useful, potentially highlighting a distinct inclusion body myositis subtype with a more severe phenotype.

Item Type: Article
Uncontrolled Keywords: all UKMYONET contributors, Cytosol, Humans, Muscle Weakness, Myositis, Inclusion Body, Electron Transport Complex IV, 5'-Nucleotidase, Autoantibodies, Prognosis, Survival Rate, Proportional Hazards Models, Retrospective Studies, Self-Help Devices, Age of Onset, Time Factors, Aged, Aged, 80 and over, Middle Aged, Female, Male, Muscle Fibers, Skeletal, Kaplan-Meier Estimate, Biomarkers
Depositing User: Symplectic Admin
Date Deposited: 25 Sep 2017 15:46
Last Modified: 19 Jan 2023 06:53
DOI: 10.1136/annrheumdis-2016-210282
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