Diagnostic potential of sarcoplasmic myxovirus resistance protein A expression in subsets of dermatomyositis

A. Uruha, Y. Allenbach, J. L. Charuel, L. Musset, A. Aussy, O. Boyer, K. Mariampillai, O. Landon-Cardinal, C. Rasmussen, L. Bolko, T. Maisonobe, S. Leonard-Louis, Shigeaki Suzuki, I. Nishino, W. Stenzel, O. Benveniste

Research output: Contribution to journalArticle

3 Citations (Scopus)

Abstract

Aims: To elucidate the diagnostic value of sarcoplasmic expression of myxovirus resistance protein A (MxA) for dermatomyositis (DM) specifically analysing different DM subforms, and to test the superiority of MxA to other markers. Methods: Immunohistochemistry for MxA and retinoic acid-inducible gene I (RIG-I) was performed on skeletal muscle samples and compared with the item presence of perifascicular atrophy (PFA) in 57 DM patients with anti-Mi-2 (n = 6), -transcription intermediary factor 1 gamma (n = 10), -nuclear matrix protein 2 (n = 13), -melanoma differentiation-associated gene 5 (MDA5) (n = 10) or -small ubiquitin-like modifier activating enzyme (n = 1) autoantibodies and with no detectable autoantibody (n = 17). Among the patients, nine suffered from cancer and 22 were juvenile-onset type. Disease controls included antisynthetase syndrome (ASS)-associated myositis (n = 30), immune-mediated necrotizing myopathy (n = 9) and inclusion body myositis (n = 5). Results: Sarcoplasmic MxA expression featured 77% sensitivity and 100% specificity for overall DM patients, while RIG-I staining and PFA reached respectively 14% and 59% sensitivity and 100% and 86% specificity. In any subset of DM, sarcoplasmic MxA expression showed higher sensitivity than RIG-I and PFA. Some anti-MDA5 antibody-positive DM samples distinctively showed a scattered staining pattern of MxA. No ASS samples had sarcoplasmic MxA expression even though six patients had DM skin rash. Conclusions: Sarcoplasmic MxA expression is more sensitive than PFA and RIG-I expression for a pathological diagnosis of DM, regardless of the autoantibody-related subgroup. In light of its high sensitivity and specificity, it may be considered a pathological hallmark of DM per se. Also, lack of MxA expression in ASS supports the idea that ASS is a distinct entity from DM.

Original languageEnglish
JournalNeuropathology and Applied Neurobiology
DOIs
Publication statusAccepted/In press - 2018 Jan 1

Fingerprint

Myxovirus Resistance Proteins
Dermatomyositis
Staphylococcal Protein A
Tretinoin
Atrophy
Autoantibodies
Genes
Melanoma
Inclusion Body Myositis
Staining and Labeling
Sensitivity and Specificity
Myositis
Muscular Diseases
Ubiquitin
Exanthema

Keywords

  • autoantibody
  • dermatomyositis
  • diagnostic marker
  • muscle pathology
  • myxovirus resistance protein A
  • type 1 interferon

ASJC Scopus subject areas

  • Pathology and Forensic Medicine
  • Histology
  • Neurology
  • Clinical Neurology
  • Physiology (medical)

Cite this

Uruha, A., Allenbach, Y., Charuel, J. L., Musset, L., Aussy, A., Boyer, O., ... Benveniste, O. (Accepted/In press). Diagnostic potential of sarcoplasmic myxovirus resistance protein A expression in subsets of dermatomyositis. Neuropathology and Applied Neurobiology. https://doi.org/10.1111/nan.12519

Diagnostic potential of sarcoplasmic myxovirus resistance protein A expression in subsets of dermatomyositis. / Uruha, A.; Allenbach, Y.; Charuel, J. L.; Musset, L.; Aussy, A.; Boyer, O.; Mariampillai, K.; Landon-Cardinal, O.; Rasmussen, C.; Bolko, L.; Maisonobe, T.; Leonard-Louis, S.; Suzuki, Shigeaki; Nishino, I.; Stenzel, W.; Benveniste, O.

In: Neuropathology and Applied Neurobiology, 01.01.2018.

Research output: Contribution to journalArticle

Uruha, A, Allenbach, Y, Charuel, JL, Musset, L, Aussy, A, Boyer, O, Mariampillai, K, Landon-Cardinal, O, Rasmussen, C, Bolko, L, Maisonobe, T, Leonard-Louis, S, Suzuki, S, Nishino, I, Stenzel, W & Benveniste, O 2018, 'Diagnostic potential of sarcoplasmic myxovirus resistance protein A expression in subsets of dermatomyositis', Neuropathology and Applied Neurobiology. https://doi.org/10.1111/nan.12519
Uruha, A. ; Allenbach, Y. ; Charuel, J. L. ; Musset, L. ; Aussy, A. ; Boyer, O. ; Mariampillai, K. ; Landon-Cardinal, O. ; Rasmussen, C. ; Bolko, L. ; Maisonobe, T. ; Leonard-Louis, S. ; Suzuki, Shigeaki ; Nishino, I. ; Stenzel, W. ; Benveniste, O. / Diagnostic potential of sarcoplasmic myxovirus resistance protein A expression in subsets of dermatomyositis. In: Neuropathology and Applied Neurobiology. 2018.
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abstract = "Aims: To elucidate the diagnostic value of sarcoplasmic expression of myxovirus resistance protein A (MxA) for dermatomyositis (DM) specifically analysing different DM subforms, and to test the superiority of MxA to other markers. Methods: Immunohistochemistry for MxA and retinoic acid-inducible gene I (RIG-I) was performed on skeletal muscle samples and compared with the item presence of perifascicular atrophy (PFA) in 57 DM patients with anti-Mi-2 (n = 6), -transcription intermediary factor 1 gamma (n = 10), -nuclear matrix protein 2 (n = 13), -melanoma differentiation-associated gene 5 (MDA5) (n = 10) or -small ubiquitin-like modifier activating enzyme (n = 1) autoantibodies and with no detectable autoantibody (n = 17). Among the patients, nine suffered from cancer and 22 were juvenile-onset type. Disease controls included antisynthetase syndrome (ASS)-associated myositis (n = 30), immune-mediated necrotizing myopathy (n = 9) and inclusion body myositis (n = 5). Results: Sarcoplasmic MxA expression featured 77{\%} sensitivity and 100{\%} specificity for overall DM patients, while RIG-I staining and PFA reached respectively 14{\%} and 59{\%} sensitivity and 100{\%} and 86{\%} specificity. In any subset of DM, sarcoplasmic MxA expression showed higher sensitivity than RIG-I and PFA. Some anti-MDA5 antibody-positive DM samples distinctively showed a scattered staining pattern of MxA. No ASS samples had sarcoplasmic MxA expression even though six patients had DM skin rash. Conclusions: Sarcoplasmic MxA expression is more sensitive than PFA and RIG-I expression for a pathological diagnosis of DM, regardless of the autoantibody-related subgroup. In light of its high sensitivity and specificity, it may be considered a pathological hallmark of DM per se. Also, lack of MxA expression in ASS supports the idea that ASS is a distinct entity from DM.",
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T1 - Diagnostic potential of sarcoplasmic myxovirus resistance protein A expression in subsets of dermatomyositis

AU - Uruha, A.

AU - Allenbach, Y.

AU - Charuel, J. L.

AU - Musset, L.

AU - Aussy, A.

AU - Boyer, O.

AU - Mariampillai, K.

AU - Landon-Cardinal, O.

AU - Rasmussen, C.

AU - Bolko, L.

AU - Maisonobe, T.

AU - Leonard-Louis, S.

AU - Suzuki, Shigeaki

AU - Nishino, I.

AU - Stenzel, W.

AU - Benveniste, O.

PY - 2018/1/1

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N2 - Aims: To elucidate the diagnostic value of sarcoplasmic expression of myxovirus resistance protein A (MxA) for dermatomyositis (DM) specifically analysing different DM subforms, and to test the superiority of MxA to other markers. Methods: Immunohistochemistry for MxA and retinoic acid-inducible gene I (RIG-I) was performed on skeletal muscle samples and compared with the item presence of perifascicular atrophy (PFA) in 57 DM patients with anti-Mi-2 (n = 6), -transcription intermediary factor 1 gamma (n = 10), -nuclear matrix protein 2 (n = 13), -melanoma differentiation-associated gene 5 (MDA5) (n = 10) or -small ubiquitin-like modifier activating enzyme (n = 1) autoantibodies and with no detectable autoantibody (n = 17). Among the patients, nine suffered from cancer and 22 were juvenile-onset type. Disease controls included antisynthetase syndrome (ASS)-associated myositis (n = 30), immune-mediated necrotizing myopathy (n = 9) and inclusion body myositis (n = 5). Results: Sarcoplasmic MxA expression featured 77% sensitivity and 100% specificity for overall DM patients, while RIG-I staining and PFA reached respectively 14% and 59% sensitivity and 100% and 86% specificity. In any subset of DM, sarcoplasmic MxA expression showed higher sensitivity than RIG-I and PFA. Some anti-MDA5 antibody-positive DM samples distinctively showed a scattered staining pattern of MxA. No ASS samples had sarcoplasmic MxA expression even though six patients had DM skin rash. Conclusions: Sarcoplasmic MxA expression is more sensitive than PFA and RIG-I expression for a pathological diagnosis of DM, regardless of the autoantibody-related subgroup. In light of its high sensitivity and specificity, it may be considered a pathological hallmark of DM per se. Also, lack of MxA expression in ASS supports the idea that ASS is a distinct entity from DM.

AB - Aims: To elucidate the diagnostic value of sarcoplasmic expression of myxovirus resistance protein A (MxA) for dermatomyositis (DM) specifically analysing different DM subforms, and to test the superiority of MxA to other markers. Methods: Immunohistochemistry for MxA and retinoic acid-inducible gene I (RIG-I) was performed on skeletal muscle samples and compared with the item presence of perifascicular atrophy (PFA) in 57 DM patients with anti-Mi-2 (n = 6), -transcription intermediary factor 1 gamma (n = 10), -nuclear matrix protein 2 (n = 13), -melanoma differentiation-associated gene 5 (MDA5) (n = 10) or -small ubiquitin-like modifier activating enzyme (n = 1) autoantibodies and with no detectable autoantibody (n = 17). Among the patients, nine suffered from cancer and 22 were juvenile-onset type. Disease controls included antisynthetase syndrome (ASS)-associated myositis (n = 30), immune-mediated necrotizing myopathy (n = 9) and inclusion body myositis (n = 5). Results: Sarcoplasmic MxA expression featured 77% sensitivity and 100% specificity for overall DM patients, while RIG-I staining and PFA reached respectively 14% and 59% sensitivity and 100% and 86% specificity. In any subset of DM, sarcoplasmic MxA expression showed higher sensitivity than RIG-I and PFA. Some anti-MDA5 antibody-positive DM samples distinctively showed a scattered staining pattern of MxA. No ASS samples had sarcoplasmic MxA expression even though six patients had DM skin rash. Conclusions: Sarcoplasmic MxA expression is more sensitive than PFA and RIG-I expression for a pathological diagnosis of DM, regardless of the autoantibody-related subgroup. In light of its high sensitivity and specificity, it may be considered a pathological hallmark of DM per se. Also, lack of MxA expression in ASS supports the idea that ASS is a distinct entity from DM.

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KW - type 1 interferon

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