More prominent muscle involvement in patients with dermatomyositis with anti-Mi2 autoantibodies.


Journal

Neurology
ISSN: 1526-632X
Titre abrégé: Neurology
Pays: United States
ID NLM: 0401060

Informations de publication

Date de publication:
05 11 2019
Historique:
received: 14 03 2019
accepted: 29 05 2019
pubmed: 9 10 2019
medline: 6 2 2020
entrez: 10 10 2019
Statut: ppublish

Résumé

To define the clinical phenotype of dermatomyositis (DM) with anti-Mi2 autoantibodies. In this longitudinal cohort study, the prevalence and severity of clinical features at disease onset and during follow-up in patients with anti-Mi2-positive DM were compared to patients with anti-Mi2-negative DM, antisynthetase syndrome (AS), and immune-mediated necrotizing myopathy (IMNM). Longitudinal anti-Mi2 autoantibody titers were assessed. A total of 58 patients with anti-Mi2-positive DM, 143 patients with anti-Mi2-negative DM, 162 patients with AS, and 170 patients with IMNM were included. Among patients with anti-Mi2-positive DM, muscle weakness was present in 60% at disease onset and occurred in 98% during longitudinal follow-up; fewer patients with anti-Mi2-negative DM developed weakness (85%; Patients with anti-Mi2-positive DM have more severe muscle disease than patients with anti-Mi2-negative DM or patients with AS. Anti-Mi2 autoantibody levels correlate with disease severity and may normalize in patients who enter remission.

Identifiants

pubmed: 31594859
pii: WNL.0000000000008443
doi: 10.1212/WNL.0000000000008443
pmc: PMC6946486
doi:

Substances chimiques

Autoantibodies 0
CHD4 protein, human 0
Creatine Kinase EC 2.7.3.2
Mi-2 Nucleosome Remodeling and Deacetylase Complex EC 3.5.1.98

Types de publication

Journal Article Research Support, N.I.H., Intramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

e1768-e1777

Informations de copyright

© 2019 American Academy of Neurology.

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Auteurs

Iago Pinal-Fernandez (I)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia. andrew.mammen@nih.gov iago.pinalfernandez@nih.gov.

Christopher A Mecoli (CA)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Maria Casal-Dominguez (M)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Katherine Pak (K)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Yuji Hosono (Y)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Julio Huapaya (J)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Wilson Huang (W)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Jemima Albayda (J)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Eleni Tiniakou (E)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Julie J Paik (JJ)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Cheilonda Johnson (C)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Sonye K Danoff (SK)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Andrea M Corse (AM)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Lisa Christopher-Stine (L)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia.

Andrew L Mammen (AL)

From the Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation (I.P.-F., M.C.-D., K.P., Y.H., W.H., A.L.M.), National Institute of Arthritis and Musculoskeletal and Skin Diseases, NIH, Bethesda; Departments of Neurology (I.P.-F., M.C.-D., A.M.C., L.C.-S., A.L.M.) and Medicine (C.A.M., J.A., E.T., J.J.P., S.K.D., L.C.-S., A.L.M.), Johns Hopkins University School of Medicine, Baltimore, MD; Faculty of Health Sciences (I.P.-F.), Universitat Oberta de Catalunya, Barcelona, Spain; Department of Medicine (J.H.), Medstar Georgetown University Hospital, Washington, DC; and Department of Medicine (C.J.), Hospital of the University of Pennsylvania, Philadelphia. andrew.mammen@nih.gov iago.pinalfernandez@nih.gov.

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