Fatal Rapidly Progressive Interstitial Pneumonitis Associated With Amyopathic Dermatomyositis and CD8 T Lymphocytes

Yokoyama, Toshinobu; Sakamoto, Teruo; Shida, Norihiko; Kinoshita, Masaharu; Kunou, Yoshifumi; Karukaya, Ritsuko; Takamatsu, Manabu; Aizawa, Hisamichi

doi:10.1177/0885066605275391

Cited by 20 publications

(18 citation statements)

References 12 publications

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“…Regarding the anti-Jo-1 antibody, only one patient with chronic ILD-ADM had this antibody among the present ILD-ADM study. To date, several studies have reported a low incidence of anti-Jo-1 antibody in ILD-ADM patients [11][12][13][17][18][19]. The current results were consistent with those studies.…”

Section: Discussionsupporting

confidence: 92%

“…To date, little is known about the BAL findings of ILD-ADM. Two reports from Japan demonstrated an increase of BALF lymphocytes and neutrophils in acute/subacute ILD-ADM [11,12]. The present study showed that the BAL findings differed between the two forms.…”

Section: Discussioncontrasting

confidence: 43%

“…As ADM is a rare disease, previous studies of ILD-ADM included only one, or a few patients [7][8][9][10][11][12][13]. The present study investigated the highest number of patients with ILD-ADM so far.…”

Section: Discussionmentioning

confidence: 98%

“…To date, contradictory data have been reported in the prognosis of ILD-ADM. Previous studies, mainly from Asia, have demonstrated that ILD-ADM generally runs an aggressive course, leading to fatal respiratory failure [7][8][9][10][11][12]. In contrast, COTTIN et al [13] recently described a benign form of ILD-ADM.…”

Section: Discussionmentioning

confidence: 99%

“…Several studies have demonstrated that rapidly progressive interstitial lung disease (ILD) with a poor prognosis occurs in patients with ADM [7][8][9][10][11][12]. These patients were often resistant to intensive therapy, such as highdose corticosteroids plus immunosuppressive agents, resulting in fatal respiratory failure.…”

mentioning

confidence: 99%

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Interstitial lung diseases associated with amyopathic dermatomyositis

Suda¹,

Fujisawa²,

Enomoto³

et al. 2006

Eur Respir J

152

116

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The aim of the present study was to clarify the clinical characteristics and prognosis of patients with interstitial lung disease (ILD) associated with amyopathic dermatomyositis (ILD-ADM).The study consisted of 14 consecutive patients with ILD-ADM. Patients were classified into two categories, acute/subacute and chronic forms, according to the clinical presentation of ILD. The clinical features, responsiveness to therapy, and prognosis between the two forms were compared.Nine ILD-ADM patients were categorised as the acute/subacute form, and five as the chronic form. Arterial oxygen tension was significantly lower in the acute/subacute ILD than chronic ILD patients. On high-resolution computed tomography, ground-glass opacities were frequently found in the two forms, but consolidation was more common in acute/subacute ILD than chronic ILD. Bronchoalveolar lavage analysis showed higher numbers of total cells and lymphocytes in acute/subacute ILD than chronic ILD. Histologically, the most common finding was nonspecific interstitial pneumonia in the two forms, while diffuse alveolar damage was only found in acute/ subacute ILD. Acute/subacute ILD was generally resistant to therapy, while chronic ILD responded well. Notably, the mortality of acute/subacute ILD was much higher than that of chronic ILD (67 versus 0%, respectively).In conclusion, interstitial lung disease associated with amyopathic dermatomyositis includes two different forms, the acute/subacute and chronic forms, with distinct prognoses.

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Section: Discussionsupporting

confidence: 92%

Section: Discussioncontrasting

confidence: 43%

Section: Discussionmentioning

confidence: 98%

Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

See 3 more Smart Citations

Interstitial lung diseases associated with amyopathic dermatomyositis

Suda¹,

Fujisawa²,

Enomoto³

et al. 2006

Eur Respir J

152

116

View full text Add to dashboard Cite

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Pneumomediastinum and subcutaneous emphysema secondary to amyopathic dermatomyositis with cryptogenic organizing pneumonia in invasive breast cancer: a case report and review of literature

et al. 2008

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Amyopathic dermatomyositis (ADM) is recognized as a variant phenotype of dermatomyositis and characterized by typical skin manifestations without evidence of muscular inflammation. While interstitial lung disease (ILD) is occasionally found as one of the lung manifestations in ADM patients, the development of a pneumomediastinum and/or subcutaneous emphysema in this disease entity is one of the extremely rare pulmonary complications. These latter complicated pulmonary manifestations have been usually reported in idiopathic ADM with ILD without any other associated medical conditions. We report a case presented with the spontaneous pneumomediastinum and subcutaneous emphysema in both ADM and cryptogenic organizing pneumonia during adjuvant chemotherapy based on cyclophosphamide for breast cancer.

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Anti-MDA5 antibody is associated with A/SIP and decreased T cells in peripheral blood and predicts poor prognosis of ILD in Chinese patients with dermatomyositis

et al. 2011

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The aim of this study is to determine serum anti-melanoma differentiation-associated gene 5 prevalence and their clinical associations in Chinese patients with polymyositis and dermatomyositis (PM/DM). Serum anti-MDA5 antibody was detected by ELISA in 113 adult PM/DM patients and in various controls. Flow cytometry was applied to analyze the subgroups of lymphocytes in the peripheral blood of PM/DM patients. The serum anti-MDA5-positive rate in the DM patients (22.6%) was significantly higher compared with that in PM patients (0%, P < 0.005), patients with SLE (3.3%, P < 0.05), RA (3.3%, P < 0.05), pSS (0%, P < 0.05), pulmonary infection (0%, P < 0.05) and healthy controls (0%, P < 0.001). The percentage of decreased CD4(+), CD8(+) T cell counts, raised CD4(+)/CD8(+) ratio in peripheral blood and the incidence of acute/subacute interstitial pneumonia (A/SIP) were significantly higher in anti-MDA5-positive DM group than negative group (all P < 0.05). Additionally, logistic multivariate analysis showed that anti-MDA5 is an independent risk factor for death of ILD in DM (OR = 8.46, 95% CI 1.77-40.36, P = 0.007). In conclusion, in Chinese PM/DM patients, serum anti-MDA5 antibody is mainly presented in DM patients and can be a useful marker for A/SIP in patients with DM. It can predict unfavorable prognosis in DM patients with ILD. Further studies are needed to identify how the abnormal T cells in peripheral blood participated in the generation of the anti-MDA5 antibody.

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Fatal Rapidly Progressive Interstitial Pneumonitis Associated With Amyopathic Dermatomyositis and CD8 T Lymphocytes

Cited by 20 publications

References 12 publications

Interstitial lung diseases associated with amyopathic dermatomyositis

Interstitial lung diseases associated with amyopathic dermatomyositis

Pneumomediastinum and subcutaneous emphysema secondary to amyopathic dermatomyositis with cryptogenic organizing pneumonia in invasive breast cancer: a case report and review of literature

Anti-MDA5 antibody is associated with A/SIP and decreased T cells in peripheral blood and predicts poor prognosis of ILD in Chinese patients with dermatomyositis

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