Anti-SRP immune-mediated necrotizing myopathy: A critical review of current concepts

Ma, Xue; Bu, Bitao

doi:10.3389/fimmu.2022.1019972

Cited by 22 publications

(7 citation statements)

References 91 publications

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“…The frequent development of RP-ILD, the scarce but fatal secondary hemophagocytic lymphohistiocytosis (sHLH), occurrence of opportunistic infection, involvement of pharyngeal and respiratory muscle, all contributed to the unfavorable outcome in these patients [ 15 , 32 – 34 ]. Previously patients with positivity of anti-SRP antibody were reported to be vulnerable to cardiac involvement [ 35 ]. In 2022, Zeng, etc.…”

Section: Discussionmentioning

confidence: 99%

An externally validated clinical-laboratory nomogram for myocardial involvement in adult idiopathic-inflammatory-myopathy patients

Liang,

Wan,

Yao

et al. 2024

Clin Rheumatol

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Objectives This study aimed at identifying clinical and laboratory risk factors for myocardial involvement (MI) in idiopathic inflammatory myopathies (IIMs) patients as well as constructing a risk-predicted nomogram for prediction and early identification of MI. Methods An IIMs cohort in southeastern China was constructed, including 504 adult IIMs patients who met the inclusion and exclusion criteria, and were hospitalized at four divisions of the First Affiliated Hospital, Zhejiang University School of Medicine from January 1st 2018 to April 30st 2022. After dividing patients into the training cohort and the validation cohort, risk factors for MI were identified through least absolute shrinkage and selection operator regression and multivariate logistic regression. A risk-predicted nomogram was established and validated internally and externally for discrimination, calibration and practicability. Results In this cohort, 17.7% of patients developed MI and the survival was significantly inferior to that of IIMs patients without MI (P < 0.001). In the training cohort, age > 55 years old (P < 0.001), disease activity > 10 points (P < 0.001), interleukin-17A (IL-17A) > 7.5 pg/ml (P < 0.001), lactic dehydrogenase (LDH) > 425 U/L (P < 0.001), anti-mitochondrial antibodies (AMAs, P = 0.017), and anti-MDA5 antibody (P = 0.037) were significantly correlated with development of MI. A nomogram was established by including the above values to predict MI and was found efficient in discrimination, calibration, and practicability through internal and external validation. Conclusion This study developed and validated a nomogram model to predict the risk of MI in adult IIMs patients, which can benefit the prediction and early identification of MI as well as timely intervention in these patients.

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Section: Discussionmentioning

confidence: 99%

An externally validated clinical-laboratory nomogram for myocardial involvement in adult idiopathic-inflammatory-myopathy patients

Liang,

Wan,

Yao

et al. 2024

Clin Rheumatol

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“…However, this association has been inconsistently reported ( 10 , 11 ), with some studies suggesting a link between the positivity for this antibody and ILD but not cardiac manifestations ( 20 ). Recent evidence suggests that anti-SRP antibodies may have a pathogenic role targeting skeletal muscle ( 21 , 22 ), with muscle fibre necrosis being confirmed in mice with transferred anti-SRP ( 23 ). Whether this contributes to cardiac involvement remains unclear.…”

Section: Discussionmentioning

confidence: 99%

Predictors of cardiac involvement in idiopathic inflammatory myopathies

Bandeira

Dourado²,

Melo³

et al. 2023

Front. Immunol.

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ObjectivesIdiopathic inflammatory myopathies (IIM) are a group of rare disorders that can affect the heart. This work aimed to find predictors of cardiac involvement in IIM.MethodsMulticenter, open cohort study, including patients registered in the IIM module of the Rheumatic Diseases Portuguese Register (Reuma.pt/Myositis) until January 2022. Patients without cardiac involvement information were excluded. Myo(peri)carditis, dilated cardiomyopathy, conduction abnormalities, and/or premature coronary artery disease were considered.Results230 patients were included, 163 (70.9%) of whom were females. Thirteen patients (5.7%) had cardiac involvement. Compared with IIM patients without cardiac involvement, these patients had a lower bilateral manual muscle testing score (MMT) at the peak of muscle weakness [108.0 ± 55.0 vs 147.5 ± 22.0, p=0.008] and more frequently had oesophageal [6/12 (50.0%) vs 33/207 (15.9%), p=0.009] and lung [10/13 (76.9%) vs 68/216 (31.5%), p=0.001] involvements. Anti-SRP antibodies were more commonly identified in patients with cardiac involvement [3/11 (27.3%) vs 9/174 (5.2%), p=0.026]. In the multivariate analysis, positivity for anti-SRP antibodies (OR 104.3, 95% CI: 2.5-4277.8, p=0.014) was a predictor of cardiac involvement, regardless of sex, ethnicity, age at diagnosis, and lung involvement. Sensitivity analysis confirmed these results.ConclusionAnti-SRP antibodies were predictors of cardiac involvement in our cohort of IIM patients, irrespective of demographical characteristics and lung involvement. We suggest considering frequent screening for heart involvement in anti-SRP-positive IIM patients.

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“…These cells play a crucial role in the induction and/or maintenance of pSS, such as providing a stimulus for B lymphocytes, and promoting the destruction of the gland through cytotoxicity [ 36 ]. Previous literatures had demonstrated that pathological characteristics of SRP-IMNM showed scattered or focal CD68 + macrophage infiltration, scattered CD4 + and CD8 + T lymphocyte, and a few CD20 + B lymphocytes, scattered CD56 + myofiber regeneration as well [ 37 ]. We hypothesized that abundant CD4 + T cells in the muscle specimens of anti-SRP IMNM associated with SS might be involved the immunological mechanism, but further mechanistic studies will be required to discuss.…”

Section: Discussionmentioning

confidence: 99%

Anti-signal recognition particle positive necrotizing myopathy-sjogren’s syndrome overlap syndrome: a descriptive study on clinical and myopathology features

Yang

et al. 2023

BMC Musculoskelet Disord

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Background and objective The aim of this study was to elucidate the clinical and myopathological characteristics of patients with anti-signal recognition particle (SRP) positive immune-mediated necrotizing myopathy (IMNM) overlap Sjogren’s syndrome (SS). Materials and methods We retrospectively analyzed the data of anti-SRP positive IMNM patients admitted in the Neurology Department of Tongji Hospital between January 2011 to December 2020. Patients were divided into two groups: anti-SRP IMNM overlap SS group and anti-SRP IMNM control group. The clinical features, laboratory results, histological features, treatment, and prognosis were compared between the two groups. Results A total of 30 patients with anti-SRP IMNM were included, including six anti-SRP IMNM overlap SS patients (two males, four females), with a median age of 39 years, and 24 anti-SRP IMNM patients (ten males, fourteen females), with a median age of 46 years. The anti-SRP IMNM overlap SS group had a lower prevalence of muscle atrophy (0 vs 50%, p = 0.019), and a higher prevalence of extramuscular manifestations, including cardiac abnormalities and ILD (Interstitial lung disease). CD4 + and CD68 + inflammatory infiltrations were significantly increased in anti-SRP IMNM overlap SS patients, with an increased presence of CD4 + cells in both necrotic(p = 0.023) and endomysial areas (p = 0.013), and more CD68 + cells (p = 0.016) infiltrated the endomysial area. Deposition of membrane attack complex (MAC) on sarcolemma (p = 0.013) was more commonly seen in the anti-SRP IMNM overlap SS group. Conclusion Our data revealed that anti-SRP IMNM-SS overlap patients may present with milder muscular manifestation, but worse extramuscular manifestations compared to anti-SRP IMNM patients without SS. CD4 + and CD68 + inflammatory infiltrations and MAC deposition were remarkably increased in anti-SRP IMNM-SS overlap patients.

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Anti-SRP immune-mediated necrotizing myopathy: A critical review of current concepts

Cited by 22 publications

References 91 publications

An externally validated clinical-laboratory nomogram for myocardial involvement in adult idiopathic-inflammatory-myopathy patients

An externally validated clinical-laboratory nomogram for myocardial involvement in adult idiopathic-inflammatory-myopathy patients

Predictors of cardiac involvement in idiopathic inflammatory myopathies

Anti-signal recognition particle positive necrotizing myopathy-sjogren’s syndrome overlap syndrome: a descriptive study on clinical and myopathology features

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