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

J B Lilleker, A Rietveld, S R Pye, K Mariampillai, O Benveniste, M T J Peeters, J A L Miller, M G Hanna, P M Machado, M J Parton, K R Gheorghe, U A Badrising, I E Lundberg, S Sacconi, M K Herbert, N J McHugh, B R F Lecky, C Brierley, D Hilton-Jones, J A Lamb & 7 others M E Roberts, R G Cooper, C G J Saris, G J M Pruijn, H Chinoy, B G M van Engelen, all UKMYONET contributors

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Abstract

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.

LanguageEnglish
JournalAnnals of the Rheumatic Diseases
Volume76
Issue number5
Early online date25 Jan 2017
DOIs
StatusPublished - 1 May 2017

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Inclusion Body Myositis
5'-Nucleotidase
Autoantibodies
Antibodies
Mortality
Biopsy
Electron Transport Complex IV
Upper Extremity
Registries
Muscle
Hazards
Retrospective Studies
Enzyme-Linked Immunosorbent Assay
Phenotype

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Lilleker, J. B., Rietveld, A., Pye, S. R., Mariampillai, K., Benveniste, O., Peeters, M. T. J., ... all UKMYONET contributors (2017). Cytosolic 5'-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis. Annals of the Rheumatic Diseases, 76(5). https://doi.org/10.1136/annrheumdis-2016-210282

Cytosolic 5'-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis. / Lilleker, J B; Rietveld, A; Pye, S R; Mariampillai, K; Benveniste, O; Peeters, M T J; Miller, J A L; Hanna, M G; Machado, P M; Parton, M J; Gheorghe, K R; Badrising, U A; Lundberg, I E; Sacconi, S; Herbert, M K; McHugh, N J; Lecky, B R F; Brierley, C; Hilton-Jones, D; Lamb, J A; Roberts, M E; Cooper, R G; Saris, C G J; Pruijn, G J M; Chinoy, H; van Engelen, B G M; all UKMYONET contributors.

In: Annals of the Rheumatic Diseases, Vol. 76, No. 5, 01.05.2017.

Research output: Contribution to journalArticle

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 & all UKMYONET contributors 2017, 'Cytosolic 5'-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis', Annals of the Rheumatic Diseases, vol. 76, no. 5. https://doi.org/10.1136/annrheumdis-2016-210282
Lilleker, J B ; Rietveld, A ; Pye, S R ; Mariampillai, K ; Benveniste, O ; Peeters, M T J ; Miller, J A L ; Hanna, M G ; Machado, P M ; Parton, M J ; Gheorghe, K R ; Badrising, U A ; Lundberg, I E ; Sacconi, S ; Herbert, M K ; McHugh, N J ; Lecky, B R F ; Brierley, C ; Hilton-Jones, D ; Lamb, J A ; Roberts, M E ; Cooper, R G ; Saris, C G J ; Pruijn, G J M ; Chinoy, H ; van Engelen, B G M ; all UKMYONET contributors. / Cytosolic 5'-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis. In: Annals of the Rheumatic Diseases. 2017 ; Vol. 76, No. 5.
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abstract = "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.",
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T1 - Cytosolic 5'-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis

AU - Lilleker, J B

AU - Rietveld, A

AU - Pye, S R

AU - Mariampillai, K

AU - Benveniste, O

AU - Peeters, M T J

AU - Miller, J A L

AU - Hanna, M G

AU - Machado, P M

AU - Parton, M J

AU - Gheorghe, K R

AU - Badrising, U A

AU - Lundberg, I E

AU - Sacconi, S

AU - Herbert, M K

AU - McHugh, N J

AU - Lecky, B R F

AU - Brierley, C

AU - Hilton-Jones, D

AU - Lamb, J A

AU - Roberts, M E

AU - Cooper, R G

AU - Saris, C G J

AU - Pruijn, G J M

AU - Chinoy, H

AU - van Engelen, B G M

AU - all UKMYONET contributors

N1 - Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing/.

PY - 2017/5/1

Y1 - 2017/5/1

N2 - 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.

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.

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

U2 - 10.1136/annrheumdis-2016-210282

DO - 10.1136/annrheumdis-2016-210282

M3 - Article

VL - 76

JO - Annals of the Rheumatic Diseases

T2 - Annals of the Rheumatic Diseases

JF - Annals of the Rheumatic Diseases

SN - 0003-4967

IS - 5

ER -