دورية أكاديمية
أعرض في EDS

A case of autoimmune pulmonary alveolar proteinosis during the course of treatment of rapidly progressive interstitial pneumonia associated with anti-MDA5 antibody-positive dermatomyositis.

التفاصيل البيبلوغرافية
العنوان: A case of autoimmune pulmonary alveolar proteinosis during the course of treatment of rapidly progressive interstitial pneumonia associated with anti-MDA5 antibody-positive dermatomyositis.
المؤلفون: Yatomi M; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan. m09702007@gunma-u.ac.jp., Akasaka K; Department of Respiratory Medicine, Saitama Red Cross Hospital, 1-5, Shintoshin, Chuo-Ku, Saitama, 330-8553, Japan., Sato S; Department of Respiratory Medicine, Saitama Red Cross Hospital, 1-5, Shintoshin, Chuo-Ku, Saitama, 330-8553, Japan., Chida M; Department of Dermatology, Gunma University Graduate School of Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Kanbe M; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Sawada H; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Yokota I; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Wakamatsu I; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Muto S; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Sato M; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Yamaguchi K; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Miura Y; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Tsurumaki H; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Sakurai R; Oncology Center, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371-, 8511, Japan., Hara K; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Koga Y; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Sunaga N; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Yamakawa H; Department of Respiratory Medicine, Saitama Red Cross Hospital, 1-5, Shintoshin, Chuo-Ku, Saitama, 330-8553, Japan., Matsushima H; Department of Respiratory Medicine, Saitama Red Cross Hospital, 1-5, Shintoshin, Chuo-Ku, Saitama, 330-8553, Japan., Yamazaki S; Department of Dermatology, Gunma University Graduate School of Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Endo Y; Department of Dermatology, Gunma University Graduate School of Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Motegi SI; Department of Dermatology, Gunma University Graduate School of Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan., Hisada T; Gunma University Graduate School of Health Sciences, 3-39-22 Showa-Machi, Maebashi, Gunma, 371-8514, Japan., Maeno T; Division of Allergy and Respiratory Medicine, Integrative Center of Internal Medicine, Gunma University Hospital, 3-39-15 Showa-Machi, Maebashi, Gunma, 371- 8511, Japan.
المصدر: BMC pulmonary medicine [BMC Pulm Med] 2024 Apr 08; Vol. 24 (1), pp. 170. Date of Electronic Publication: 2024 Apr 08.
نوع المنشور: Case Reports; Journal Article
اللغة: English
بيانات الدورية: Publisher: BioMed Central Country of Publication: England NLM ID: 100968563 Publication Model: Electronic Cited Medium: Internet ISSN: 1471-2466 (Electronic) Linking ISSN: 14712466 NLM ISO Abbreviation: BMC Pulm Med Subsets: MEDLINE
أسماء مطبوعة: Original Publication: London : BioMed Central, [2001]-
مواضيع طبية MeSH: Pulmonary Alveolar Proteinosis*/complications , Pulmonary Alveolar Proteinosis*/diagnosis , Pulmonary Alveolar Proteinosis*/drug therapy , Dermatomyositis*/complications , Dermatomyositis*/drug therapy , Lung Diseases, Interstitial*/complications , Lung Diseases, Interstitial*/diagnosis , Lung Diseases, Interstitial*/drug therapy , Dermatitis*/complications , Autoimmune Diseases*, Female ; Humans ; Middle Aged ; Autoantibodies ; Cyclophosphamide/therapeutic use ; Interferon-Induced Helicase, IFIH1
مستخلص: Background: Autoimmune pulmonary alveolar proteinosis (APAP) is a diffuse lung disease that causes abnormal accumulation of lipoproteins in the alveoli; however, its pathogenesis remains unclear. Recently, APAP cases have been reported during the course of dermatomyositis. The combination of these two diseases may be coincidental; however, it may have been overlooked because differentiating APAP from a flare-up of interstitial pneumonia associated with dermatomyositis is challenging. This didactic case demonstrates the need for early APAP scrutiny.
Case Presentation: A 50-year-old woman was diagnosed with anti-melanoma differentiation-associated gene 5 (anti-MDA5) antibody-positive dermatitis and interstitial pneumonia in April 2021. The patient was treated with corticosteroids, tacrolimus, and cyclophosphamide pulse therapy for interstitial pneumonia complicated by MDA5 antibody-positive dermatitis, which improved the symptoms and interstitial pneumonia. Eight months after the start of treatment, a new interstitial shadow appeared that worsened. Therefore, three additional courses of cyclophosphamide pulse therapy were administered; however, the respiratory symptoms and interstitial shadows did not improve. Respiratory failure progressed, and 14 months after treatment initiation, bronchoscopy revealed turbid alveolar lavage fluid, numerous foamy macrophages, and numerous periodic acid-Schiff-positive unstructured materials. Blood test results revealed high anti-granulocyte-macrophage colony-stimulating factor (GM-CSF) antibody levels, leading to a diagnosis of APAP. The patient underwent whole-lung lavage, and the respiratory disturbance promptly improved. Anti-GM-CSF antibodies were measured from the cryopreserved serum samples collected at the time of diagnosis of anti-MDA5 antibody-positive dermatitis, and 10 months later, both values were significantly higher than normal.
Conclusions: This is the first report of anti-MDA5 antibody-positive dermatomyositis complicated by interstitial pneumonia with APAP, which may develop during immunosuppressive therapy and be misdiagnosed as a re-exacerbation of interstitial pneumonia. In anti-MDA5 antibody-positive dermatomyositis, APAP comorbidity may have been overlooked, and early evaluation with bronchoalveolar lavage fluid and anti-GM-CSF antibody measurements should be considered, keeping the development of APAP in mind.
(© 2024. The Author(s).)
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فهرسة مساهمة: Keywords: Autoimmune pulmonary alveolar proteinosis; Granulocyte–macrophage colony-stimulating factor; Granulocyte–macrophage colony-stimulating factor antibodies; Melanoma differentiation-associated gene 5; Rapidly progressive interstitial lung disease
المشرفين على المادة: 0 (Autoantibodies)
8N3DW7272P (Cyclophosphamide)
EC 3.6.4.13 (Interferon-Induced Helicase, IFIH1)
SCR Disease Name: Pulmonary Alveolar Proteinosis, Acquired
تواريخ الأحداث: Date Created: 20240408 Date Completed: 20240410 Latest Revision: 20240411
رمز التحديث: 20240411
مُعرف محوري في PubMed: PMC11003183
DOI: 10.1186/s12890-024-02989-9
PMID: 38589870
قاعدة البيانات: MEDLINE
الوصف
تدمد:1471-2466
DOI:10.1186/s12890-024-02989-9