Mortality factors in idiopathic inflammatory myopathy: focusing on malignancy and interstitial lung disease

Woo, Jin-Hyun; Kim, Yun Jung; Kim, Jin Ju; Choi, Chan-Bum; Sung, Yoon‐Kyoung; Kim, Tae‐Hwan; Jun, Jae‐Bum; Bae, Sang‐Cheol; Yoo, Dae-Hyun

doi:10.1007/s10165-012-0673-2

Cited by 16 publications

(25 citation statements)

References 22 publications

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“…Of the 13 patients with known cause of death, it was the primary cause for 7 patients and an associated cause in 2 patients. This is consistent with findings from adult IIM studies (6, 12, 13). Only 3 of these patients had an anti-aminoacyl-tRNA synthetase autoantibody, suggesting that in JIIM there are additional factors contributing to the development of pulmonary disease.…”

Section: Discussionsupporting

confidence: 92%

“…Random forests and RSF have been similarly used successfully by other investigators to determine mortality risk factors for other rheumatic illnesses, cardiovascular disease and malignancies (23, 26–28). Some of the risk factors that we identified, including clinical subgroup and ILD, are similar to those found in studies of adult IIM (6, 12–14). …”

Section: Discussionsupporting

confidence: 79%

“…Of the mortality features from our univariable analyses, those also described in adult IIM studies included older age at illness onset (4–6, 10–13, 29), delay in diagnosis or treatment (4, 5, 10), ILD (6, 12–14), anti-synthetase autoantibodies (8, 9, 30) and dysphagia (6). Our study in JIIM did not identify other important mortality risk factors that have been seen in adult IIM, such as malignancy (4, 5, 10–13), cardiac involvement and ischemic cardiovascular disease (6, 8, 11), skin ulcers (10, 13) and male gender (6, 11, 29).…”

Section: Discussionmentioning

confidence: 99%

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Early Illness Features Associated With Mortality in the Juvenile Idiopathic Inflammatory Myopathies

Huber

Mamyrova

Lachenbruch

et al. 2014

Arthritis Care & Research

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Objectives Because juvenile idiopathic inflammatory myopathies (JIIM) are potentially life-threatening systemic autoimmune diseases, we examined risk factors for JIIM mortality. Methods Mortality status was available for 405 patients (329 juvenile dermatomyositis [JDM], 30 juvenile polymyositis [JPM], 46 juvenile connective tissue disease–associated myositis [JCTM]) enrolled in nationwide protocols. Standardized mortality ratios (SMR) were calculated using United States population statistics. Cox regression was used to assess univariable associations with mortality, and random survival forest (RSF) classification and Cox regression for multivariable associations. Results Of 17 deaths (4.2% overall mortality), 8 (2.4%) were in JDM patients. Death was related to the pulmonary system, primarily interstitial lung disease (ILD), in 7 patients, gastrointestinal in 3, multisystem in 3, and of unknown etiology in 4 patients. The SMR for JIIM overall was 14.4 [95% confidence interval (CI) 12.2, 16.5] and 8.3 [95% CI 6.4, 10.3] for JDM. The top mortality risk factors in the univariable analysis included clinical subgroup (JCTM, JPM), anti-synthetase autoantibodies, older age at diagnosis, ILD and Raynaud’s phenomenon at diagnosis. In multivariable analyses, clinical subgroup, illness severity at onset, age at diagnosis, weight loss and delay to diagnosis were the most important predictors from RSF; clinical subgroup and illness severity at onset were confirmed by multivariable Cox regression. Conclusions Overall mortality was higher in JIIM patients, and several early illness features were identified as risk factors. Clinical subgroup, anti-synthetase autoantibodies, older age at diagnosis, and ILD are also recognized as mortality risk factors in adult myositis.

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Section: Discussionsupporting

confidence: 92%

Section: Discussionsupporting

confidence: 79%

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Early Illness Features Associated With Mortality in the Juvenile Idiopathic Inflammatory Myopathies

Huber

Mamyrova

Lachenbruch

et al. 2014

Arthritis Care & Research

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“…16,42 However, the prevalence of organ involvement (pulmonary, articular, gastrointestinal, and cardiac) was lower compared with other series. 43,44 The results of our study reveal that survival of patients with DM is compromised early on after the diagnosis of DM. This information is clinically and epidemiologically relevant.…”

Section: Discussionmentioning

confidence: 93%

Clinical and Prognostic Factors Associated With Survival in Mexican Patients With Idiopathic Inflammatory Myopathies

Galindo-Feria

Rojas-Serrano

Hinojosa‐Azaola

2016

JCR: Journal of Clinical Rheumatology

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“…Cardiac involvement, interstitial lung disease and cancer are three major causes of mortality in patients with IIM [64][65][66][67]. We showed previously how certain MSA and/or MAA are strongly associated with cardiac (e.g., anti-SRP, anti-mitochondrial antibodies) and/or lung diseases (anti-synthetases, anti-MDA5, but also anti-Ku [68] and anti-PM ScL antibodies [44] see Tables 1 and 2).…”

Section: Prognosismentioning

confidence: 99%

Diagnostic Utility of Auto-Antibodies in Inflammatory Muscle Diseases

Allenbach

Benveniste

2015

Journal of Neuromuscular Diseases

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To date, there are four main groups of idiopathic inflammatory myopathies (IIM): polymyositis (PM), dermatomyositis (DM), immune-mediated necrotizing myopathy (IMNM) and sporadic inclusion body myositis; based on clinical presentation and muscle pathology. Nevertheless, important phenotypical differences (either muscular and/or extra-muscular manifestations) within a group persist. In recent years, the titration of different myositis-specific (or associated) auto-antibodies as a diagnostic tool has increased. This is an important step forward since it may facilitate, at a viable cost, the differential diagnosis between IIM and other myopathies. We have now routine access to assays for the detection of different antibodies. For example, IMNM are related to the presence of anti-SRP or anti-HMGCR. PM is associated with anti-synthetase antibodies (anti-Jo-1, PL-7, PL-12, OJ, and EJ) and DM with anti-Mi-2, anti-SAE, anti-TIF-1-␥ and anti-NXP2 (both associated with cancer) or anti-MDA5 antibodies (associated with interstitial lung disease). Today, over 30 myositis specific and associated antibodies have been characterised, and all groups of myositis may present one of those auto-antibodies. Most of them allow identification of homogenous patient groups, more precisely than the classical international classifications of myositis. This implies that classification criteria could be modified accordingly, since these auto-antibodies delineate groups of patients suffering from myositis with consistent clinical phenotype (muscular and extra-muscular manifestations), common prognostic (cancer association, presence of interstitial lung disease, mortality and risk of relapse) and treatment responses. Nevertheless, since numerous auto-antibodies have been recently characterised, the exact prevalence of myositis specific antibodies remains to be documented, and research of new auto-antibodies in the remaining seronegative group is still needed.

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Mortality factors in idiopathic inflammatory myopathy: focusing on malignancy and interstitial lung disease

Cited by 16 publications

References 22 publications

Early Illness Features Associated With Mortality in the Juvenile Idiopathic Inflammatory Myopathies

Early Illness Features Associated With Mortality in the Juvenile Idiopathic Inflammatory Myopathies

Clinical and Prognostic Factors Associated With Survival in Mexican Patients With Idiopathic Inflammatory Myopathies

Diagnostic Utility of Auto-Antibodies in Inflammatory Muscle Diseases

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