A Systematic Review and Meta-Analysis of Prevalence Studies of Sporadic Inclusion Body Myositis

Callan, Aoife; Capkun, Gorana; Vasanthaprasad, Vijayalakshmi; Freitas, Rita; Needham, Merrilee

doi:10.3233/jnd-160198

Cited by 54 publications

(44 citation statements)

References 23 publications

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“…[1][2][3] Results from a systematic review and meta-analysis in people of all age categories showed that the pooled metaprevalence of IBM was 24•8 per million (95% confidence interval [CI], 20•0-29•6), when limited to the highest quality prevalence papers (data from nine articles). 4 IBM affects men more often than women (ratio: 2:1 to 3:1). 5 The progression of leg weakness leads to frequent falls 6 and results in the loss of ambulation, leading to the use of assistive devices for mobility and eventual wheelchair dependence.…”

Section: Introduction (3598 Words; Limit 4500)mentioning

confidence: 99%

Safety and efficacy of intravenous bimagrumab in inclusion body myositis (RESILIENT): a randomised, double-blind, placebo-controlled phase 2b trial

Hanna

Badrising

Benveniste

et al. 2019

The Lancet Neurology

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102

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Background: To assess the efficacy, safety, and tolerability of bimagrumab (fully human monoclonal antibody) in participants with inclusion body myositis (IBM). Methods: This multicentre, double-blind, placebo-controlled study (RESILIENT; ClinicalTrials.gov, number NCT01925209) was conducted between September 26, 2013 and January 06, 2016 at academic clinical sites in Europe, the USA, Australia, and Japan. Eligible participants (aged 36-85 years [inclusive]; modified 2010 MRC criteria) were randomly assigned (1:1:1:1) using blocked randomisation schedule (block size=4) to receive intravenous infusions of bimagrumab 10, 3, 1 mg/kg, or placebo every 4 weeks for at least 48 weeks. All study participants, sponsor, investigators, site personnel, and those performing assessments were masked to treatment assignment. 6-minute walking distance (6MWD; primary outcome measure) was assessed at Week 52 in the primary analysis population. A multivariate normal repeated measures model was used to analyse data on 6MWD. Safety was assessed by recording adverse events (AEs), electrocardiography, echocardiography, hematology, urinalysis, and blood chemistry.

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Section: Introduction (3598 Words; Limit 4500)mentioning

confidence: 99%

Safety and efficacy of intravenous bimagrumab in inclusion body myositis (RESILIENT): a randomised, double-blind, placebo-controlled phase 2b trial

Hanna

Badrising

Benveniste

et al. 2019

The Lancet Neurology

Self Cite

102

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“…The prevalence of sIBM is thought to range from 4.9 to 33 per million and up to 51.3 per million in those above the age of 50 (1). However, in the absence of a reliable diagnostic biomarker, the prevalence is suspected to be underestimated due to diagnostic challenges in differentiating sIBM from other IIMs (3). Previous reports have suggested diagnostic criteria for sIBM, although the highest diagnostic sensitivity and specificity requires a combination of clinical, electrodiagnostic and pathological assessment (1, 2).…”

Section: Introductionmentioning

confidence: 99%

Anti-NT5c1A Autoantibodies as Biomarkers in Inclusion Body Myositis

et al. 2019

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Objective: Sporadic Inclusion Body Myositis (sIBM) is an inflammatory myopathy (IIM) without a specific diagnostic biomarker until autoantibodies to the cytosolic 5′-nucleotidase 1A ( NT5c1A /Mup44) were reported. The objectives of our study were to determine the sensitivity and specificity of anti-NT5c1A for sIBM, demonstrate demographic, clinical and serological predictors for anti-NT5c1A positivity and determine if anti-nuclear antibody (ANA) indirect immunofluorescence (IIF) staining on HEp-2 cells is a reliable screening method for anti-NT5c1A. Methods: Sera from sIBM patients and controls were stored at −80°C until required for analysis. IgG antibodies to NT5c1A were detected by an addressable laser bead immunoassay (ALBIA) using a full-length human recombinant protein. Autoantibodies to other autoimmune myopathy antigens (Jo-1, OJ, TIF1y, PL-12, SAE, EJ, MDA5, PL7, SRP, NXP2, MI-2) were detected by line immunoassay (LIA), chemiluminescence immunoassay (CIA) or enzyme linked immunosorbent assay (ELISA) and ANA detected by IIF on HEp-2 substrate. Demographic, clinical and serological data were obtained by chart review. Results: Forty-three patients with sIBM, 537 disease control patients with other autoimmune, degenerative and neuromuscular diseases, and 78 healthy controls were included. 48.8% (21/43) of sIBM patients were positive for anti-NT5c1A. The overall sensitivity, specificity, positive predictive value, and negative predictive value of anti-NT5c1A for sIBM were 0.49, 0.92, 0.29, and 0.96, respectively. Compared to sIBM, the frequency of anti-NT5c1A was lower in both the disease control group (8.8%, OR 0.10 [95%CI: 0.05–0.20], p < 0.0001) and in the apparently healthy control group (5.1%, OR 0.06 [95%CI: 0.02–0.18], p < 0.0001). In the univariable analysis, sIBM patients with more severe muscle weakness were more likely to be anti-NT5c1A positive (OR 4.10 [95% CI: 1.17, 14.33], p = 0.027), although this was not statistically significant (adjusted OR 4.30 [95% CI: 0.89, 20.76], p = 0.069) in the multivariable analysis. The ANA of sIBM sera did not demonstrate a consistent IIF pattern associated with anti-NT5c1A. Conclusions: Anti-NT5c1A has moderate sensitivity and high specificity for sIBM using ALBIA. The presence of anti-NT5c1A antibodies may be associated with muscle weakness. Anti-NT5c1A antibodies were not associated with a specific IIF staining pattern, hence screening using HEp-2 substrate is unlikely to be a useful predictor for presence of these autoantibodies.

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“…The Methodological Evaluation of Observational Research (MORE) checklist [21] was used to verify methodological quality and risk of bias. This checklist was designed for quality and bias assessment in incidence or prevalence studies of chronic diseases, and was previously used in several systematic reviews [22][23][24][25][26][27][28]. Two authors (M.C.B., H.F.L.)…”

Section: Data Extractionmentioning

confidence: 99%

Incidence and Prevalence of Chronic Inflammatory Demyelinating Polyradiculoneuropathy: A Systematic Review and Meta-Analysis

et al. 2019

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Background: Prevalence and incidence rates of chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) are required to determine the impact of CIDP on society. We aimed to estimate the prevalence and incidence of CIDP worldwide and to determine the effect of diagnostic criteria on prevalence and incidence. Method: A systematic review was conducted for all published incidence and prevalence studies on CIDP until May 18, 2017. Methodological quality was assessed using the Methodological Evaluation of Observational Research checklist. We performed a random effect meta-analysis to estimate pooled prevalence and incidence rates. Results: Of the 907 studies, 11 were included in the systematic review, 5 in the meta-analysis of incidence (818 cases; 220,513,514 person-years) and 9 in the meta-analysis of prevalence (3,160 cases; 160,765,325 population). These studies had a moderate quality. The pooled crude incidence rate was 0.33 per 100,000 person-years (95% CI 0.21–0.53; I² = 95.7%) and the pooled prevalence rate was 2.81 per 100,000 (95% CI 1.58–4.39; I² = 99.1%). Substantial heterogeneity in incidence and prevalence across studies seems to be partly explained by using different diagnostic criteria. Conclusion: These findings provide a starting point to estimate the social burden of CIDP and demonstrate the need to reach consensus on diagnostic criteria for CIDP.

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A Systematic Review and Meta-Analysis of Prevalence Studies of Sporadic Inclusion Body Myositis

Cited by 54 publications

References 23 publications

Safety and efficacy of intravenous bimagrumab in inclusion body myositis (RESILIENT): a randomised, double-blind, placebo-controlled phase 2b trial

Safety and efficacy of intravenous bimagrumab in inclusion body myositis (RESILIENT): a randomised, double-blind, placebo-controlled phase 2b trial

Anti-NT5c1A Autoantibodies as Biomarkers in Inclusion Body Myositis

Incidence and Prevalence of Chronic Inflammatory Demyelinating Polyradiculoneuropathy: A Systematic Review and Meta-Analysis

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