Association of distinct clinical subsets with myositis‐specific autoantibodies towards anti‐155/140‐kDa polypeptides, anti‐140‐kDa polypeptides, and anti‐aminoacyl tRNA synthetases in Japanese patients with dermatomyositis: a single‐centre, cross‐sectional study

Fujikawa, Keita; Kawakami, Atsushi; Kaji, Kenzo; Fujimoto, Manabu; Kawashiri, Shin‐ya; Iwamoto, Naoki; Aramaki, Toshiyuki; Ichinose, Kunihiro; Tamai, Mami; Kamachi, Makoto; Nakamura, Hideki; Ida, Hiroaki; Origuchi, Tomoki; Ishimoto, Hiroshi; Mukae, Hiroshi; Kuwana, Masataka; Kohno, Shigeru; Takehara, Kazuhiko; Sato, Shinichi; Eguchi, Katsumi

doi:10.1080/03009740802687455

Cited by 80 publications

(94 citation statements)

References 11 publications

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“…MDA5-associated DM has been reported in Asia and in the USA [3,4,5,8,9,10,11,13,19,25,26,27,28,29,30,31]. This is the first report of such a condition in Europe.…”

Section: Discussionmentioning

confidence: 73%

“…Of interest, the rate of ILD is particularly high in CADM patients, with a more severe, corticoresistant, potentially fatal, rapidly progressive course [7]. The anti-MDA5 specificity appears to be associated with a prevalence of ILD variably reported between 22 and 93% [3,5,8,9,10,11] and with a high mortality, with death rates around 70% [10,12]. In this respect, luckily, our patient responded to a regimen of high-dose steroids and pulsed intravenous cyclophosphamide.…”

Section: Discussionmentioning

confidence: 99%

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Response of Mucocutaneous Lesions to Rituximab in a Case of Melanoma Differentiation Antigen 5-Related Dermatomyositis

Clottu¹,

Laffitte²,

Prins³

et al. 2012

Dermatology

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We report the first case in Western Europe of a person presenting with dermatomyositis associated with melanoma differentiation antigen 5 antibodies. She sequentially developed severe mucocutaneous erythematous and itchy lesions of the face, scalp, neck, knees and recurrent aphthae. In addition she presented painful, periungual, edematous digital lesions and small ulcers with digital necrosis. Her rapidly evolving, near-fatal interstitial lung disease responded to high-dose intravenous cyclophosphamide. However, her recurrent mucocutaneous mani-festations improved only after rituximab administration.

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“…MDA5-associated DM has been reported in Asia and in the USA [3,4,5,8,9,10,11,13,19,25,26,27,28,29,30,31]. This is the first report of such a condition in Europe.…”

Section: Discussionmentioning

confidence: 73%

Section: Discussionmentioning

confidence: 99%

Response of Mucocutaneous Lesions to Rituximab in a Case of Melanoma Differentiation Antigen 5-Related Dermatomyositis

Clottu¹,

Laffitte²,

Prins³

et al. 2012

Dermatology

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“…In association with different autoantibodies, these subsets vary in rates of skin involvement, muscle involvement, ILD, and underlying malignancy ( Table 1). [3][4][5] Although many of these antibodies are not yet available for routine clinical testing, knowledge of the clinical variants can assist clinicians with the diagnosis of dermatomyositis in patients who present without classic features. Anti-MDA5-positive-dermatomyositis (anti-MDA5-positive-DM) was first described in a group of patients who lacked clinical signs of muscle involvement (clinically amyopathic dermatomyositis); however, subsequent studies have also found patients with this antibody who have classic muscle involvement with weakness and elevated muscle enzymes.…”

Section: Discussionmentioning

confidence: 99%

Anti-MDA5-Positive Dermatomyositis Presenting as Fever of Unknown Origin

et al. 2016

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Dermatomyositis is a chronic systemic autoimmune disease characterized by inflammatory infiltrates in the skin and muscle. The wide variability in clinical and serologic presentation poses a diagnostic challenge for the internist. Appreciation of the clinical variants of dermatomyositis allows for expedient diagnosis and avoidance of diagnostic error. We illustrate these challenges with the case of a 51-year-old VietnameseAmerican man who initially presented with fever of unknown origin in the absence of overt skin and muscle manifestations. The diagnosis of dermatomyositis was not evident on several clinical encounters due to the absence of these hallmark symptoms. We review the variable clinical manifestations of a subtype of dermatomyositis associated with an autoantibody against melanoma differentiation-associated protein 5 (anti-MDA5) and suggest consideration of dermatomyositis as a diagnosis in patients presenting with systemic illness and markedly elevated ferritin, even in the absence of elevated muscle enzymes and classic autoantibodies. CASE PRESENTATIONA 51-year-old Vietnamese-American male industrial engineer was admitted to the medical ward for evaluation of fever of unknown origin (FUO). He reported 3 weeks of fever, fatigue, generalized weakness, and dyspnea. His past medical history included a remote 10-pack-year history of cigarette smoking and latent tuberculosis treated with 9 months of isoniazid treatment, completed 5 years prior. He had moved from Vietnam to the United States 30 years earlier and denied any recent travel, sick contacts, or insect bites. Physical examination revealed a fatigued man, with a temperature of 38.1°C, pulse of 108 beats/minute, respiratory rate of 28 breaths/minute, and room air oxygen saturation of 96 %. Oropharyngeal, neurologic, pulmonary, cardiac, musculoskeletal, and skin evaluation were otherwise normal.A complete blood count (CBC) revealed anemia (hemoglobin 12.3 g/dL, hematocrit 38.5 %). White blood cell counts, platelet counts, and the basic metabolic panel were normal. Erythrocyte sedimentation rate (ESR) was 97 mm/hr and C-reactive protein (CRP) was 5.7 mg/dL. Muscle enzymes, sent to evaluate his weakness in the setting of elevated inflammatory markers, showed normal creatine kinase (CK) at 44 units/liter and mildly elevated aldolase to 9.4 U/L (normal: < 7.7 U/L). Blood, urine, and sputum cultures had no growth. Chest x-ray (CXR) demonstrated low lung volumes with increased reticular markings, small bilateral pleural effusions, and bibasilar opacities. During the hospitalization, he continued to have intermittent fevers as high as 39.1°C, generalized weakness, and dyspnea. Additionally, he developed bibasilar crackles on pulmonary exam and flat, diffuse, slightly hyperpigmented patches on his arms and chest that faded over several days. Extensive workup for infectious, inflammatory, and malignant causes of fever was unrevealing, including negative viral hepatitis serologies, human immunodeficiency virus antibody, anti-nuclear antibody (ANA), and anti-...

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“…Patients with hypomyopathic DM had DM rash and subclinical evidence of myositis on electrophysiologic, radiologic, or laboratory evaluation. 24 Thirteen patients (5 with anti-155/140 Abs and 8 with anti-CADM-140 Abs) who were observed at Nagasaki University 25 were included in this study. As controls, serum samples from 34 patients with PM, 326 with systemic sclerosis, and 97 with systemic lupus erythematosus who were observed during the same period were also assessed.…”

Section: Patients and Serum Samplesmentioning

confidence: 99%

Clinical Correlations With Dermatomyositis-Specific Autoantibodies in Adult Japanese Patients With Dermatomyositis

Hamaguchi¹,

Kuwana²,

Hoshino³

et al. 2011

Arch Dermatol

297

258

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Association of distinct clinical subsets with myositis‐specific autoantibodies towards anti‐155/140‐kDa polypeptides, anti‐140‐kDa polypeptides, and anti‐aminoacyl tRNA synthetases in Japanese patients with dermatomyositis: a single‐centre, cross‐sectional study

Cited by 80 publications

References 11 publications

Response of Mucocutaneous Lesions to Rituximab in a Case of Melanoma Differentiation Antigen 5-Related Dermatomyositis

Response of Mucocutaneous Lesions to Rituximab in a Case of Melanoma Differentiation Antigen 5-Related Dermatomyositis

Anti-MDA5-Positive Dermatomyositis Presenting as Fever of Unknown Origin

Clinical Correlations With Dermatomyositis-Specific Autoantibodies in Adult Japanese Patients With Dermatomyositis

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