RT Journal Article
SR Electronic
T1 Cytosolic 5′-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis
JF Annals of the Rheumatic Diseases
JO Ann Rheum Dis
FD BMJ Publishing Group Ltd and European League Against Rheumatism
SP 862
OP 868
DO 10.1136/annrheumdis-2016-210282
VO 76
IS 5
A1 J B Lilleker
A1 A Rietveld
A1 S R Pye
A1 K Mariampillai
A1 O Benveniste
A1 M T J Peeters
A1 J A L Miller
A1 M G Hanna
A1 P M Machado
A1 M J Parton
A1 K R Gheorghe
A1 U A Badrising
A1 I E Lundberg
A1 S Sacconi
A1 M K Herbert
A1 N J McHugh
A1 B R F Lecky
A1 C Brierley
A1 D Hilton-Jones
A1 J A Lamb
A1 M E Roberts
A1 R G Cooper
A1 C G J Saris
A1 G J M Pruijn
A1 H Chinoy
A1 B G M van Engelen
YR 2017
UL http://ard.bmj.com/content/76/5/862.abstract
AB 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.