Evidence map›Paper›PMID 36690456›Full record

ArticleNeurology2023

Clinical Subgroups and Factors Associated With Progression in Patients With Inclusion Body Myositis.

Elizabeth Harlan Michelle, Iago Pinal-Fernandez, Maria Casal-Dominguez, Jemima Albayda, Julie J Paik, Eleni Tiniakou, Brittany Adler, Christopher A Mecoli, Sonye K Danoff, Lisa Christopher-Stine and 2 more

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In one paragraph

Article in Neurology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

13 citing papers in PubMed.

  1. Article
  2. Article
  3. Review
  4. Article
  5. Epidemiology of myositis.Current opinion in rheumatology · 2025
    Review
  6. Article
  7. Current biomarkers in inclusion body myositis.Journal of neuromuscular diseases · 2024
    Review
  8. Article
  9. Article
  10. Article
  11. Article
  12. Article
  13. Clinical pearls and promising therapies in myositis.Expert review of clinical immunology
    Review
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

12 authors.

Elizabeth Harlan MichelleFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Iago Pinal-FernandezFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Maria Casal-DominguezFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Jemima AlbaydaFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Julie J PaikFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Eleni TiniakouFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Brittany AdlerFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Christopher A MecoliFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Sonye K DanoffFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Lisa Christopher-StineFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Andrew L MammenFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain.
Thomas E LloydFrom the Departments of Neurology (E.H.M., I.P.-F., M.C.-D., A.L.M., T.E.L.), and Medicine (J.A., J.J.P., E.T., B.A., C.A.M., S.K.D., L.C.-S.), Johns Hopkins University School of Medicine, Baltimore, MD; Muscle Disease Unit (I.P.-F., M.C.-D., A.L.M.), Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD; and Faculty of Health Sciences and Faculty of Computer Science (I.P.-F.), Multimedia and Telecommunications, Universitat Oberta de Catalunya, Barcelona, Spain. tlloyd4@jhmi.edu.

Funding

Muscle Disease UnitZIAAR041203 · NIAMS · NATIONAL INSTITUTE OF ARTHRITIS AND MUSCULOSKELETAL AND SKIN DISEASES · PI MAMMEN, ANDREW · 2015 to 2025
$19.7M
Pathogenesis and treatment of sporadic Inclusion Body Myositis in mouse models.R01AR076390 · NIAMS · JOHNS HOPKINS UNIVERSITY · PI LLOYD, THOMAS E., WONG, PHILIP C · 2020 to 2024
$2.2M
RNA: Polymerase III specific CD8+ T cells: a mechanistic insight into cancer-induced autoimmunity in sclerodermaK08AR077732 · NIAMS · UNIVERSITY OF TEXAS HLTH SCI CTR HOUSTON · PI TINIAKOU, ELENI · 2021 to 2025
$794k
Skeletal Myopathy in Systemic Sclerosis: Devising a Classification Schema Predictive of OutcomesK23AR073927 · NIAMS · JOHNS HOPKINS UNIVERSITY · PI PAIK, JULIE JISUN · 2020 to 2024
$787k
NIAMS NIH HHS K08 AR077732NIAMS NIH HHS K23 AR073927NIAMS NIH HHS R01 AR076390
6 · The paper itself

Abstract

BACKGROUND AND

objectivesSporadic inclusion body myositis (IBM) is the most common acquired myopathy in individuals older than 50 years. The disorder is slowly progressive, and although many therapies have been investigated, response has generally been poor. Clinical heterogeneity may influence treatment responsiveness; however, data regarding heterogeneity in IBM are limited and often conflicting. We aim to identify clinically distinct subgroups within a large IBM cohort and prognostic factors for disease progression.

methodsClinical, histologic, radiologic, and electrophysiologic data were analyzed for all patients with IBM and other forms of myositis enrolled in a longitudinal cohort from The Johns Hopkins Myositis Center from 2003 to 2018. Patients with IBM were included if they met at least one of the following criteria: Griggs possible, European Neuromuscular Centre 2011 probable, or Lloyd-Greenberg data-derived criteria for IBM. Univariate, multivariate, and graphical analyses were used to identify prognostic factors in patients with IBM. Thus, linear and logistic regressions were used to adjust for potential confounding variables. The evolution of creatine kinase and muscle strength was studied using multilevel linear regression models. Nonmodifiable risk factors (sex, race, disease duration, and age at the onset of first symptoms) were used as adjusting covariates for the regression analyses.

resultsAmong the 335 patients meeting the inclusion criteria for IBM, 64% were male with an average age of disease onset of 58.7 years and delay to diagnosis of 5.2 years. Initial misdiagnosis (52%) and immunosuppressant treatment (42%) were common. Less than half (43%) of muscle biopsies demonstrated all 3 pathologic hallmarks: endomysial inflammation, mononuclear cell invasion, and rimmed vacuoles. Black patients had significantly weaker arm abductors, hip flexors, and knee flexors compared with non-Black patients. Female patients had stronger finger flexors and knee extensors compared with their male counterparts. Younger age (<50 years) at onset was not associated with increased weakness. DISCUSSION: Our study demonstrates that female and Black patients have distinct clinical phenotypes and trajectories within the overarching IBM clinical phenotype. These subgroups may have different responses to therapies, which may influence the design of future clinical trials in IBM.

Indexed as

MyositisMyositis, Inclusion BodyFemaleHumansImmunosuppressive AgentsInflammationMaleMuscle StrengthImmunosuppressive Agents

Identifiers

PMID36690456
PMCPMC10065210

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