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Chest CT revealed peripheral ground-glass opacities, and connective tissue disease-associated ILD was suspected. During the bronchoscopy examination, we unexpectedly observed severe laryngeal oedema, which necessitated urgent tracheal intubation. We thought it had developed suddenly, with no symptoms until just before. Following hospital admission, she was confirmed to be positive for anti-MDA5 antibody. Her ILD progressed despite appropriate treatment, and she ultimately died. An autopsy revealed localised laryngeal oedema with histiocyte infiltration, which suggested laryngeal oedema related to anti-MDA5-positive dermatomyositis. This case report indicates that laryngeal oedema may occur in these patients and rapidly progress to a severe condition. Therefore, it may be necessary to evaluate the risk of airway compromise.<\/jats:p>","DOI":"10.1136\/bcr-2025-266696","type":"journal-article","created":{"date-parts":[[2025,8,12]],"date-time":"2025-08-12T02:30:34Z","timestamp":1754965834000},"page":"e266696","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Severe laryngeal oedema unexpectedly observed during bronchoscopy in a patient with anti-MDA5-positive dermatomyositis"],"prefix":"10.1136","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-7636-6255","authenticated-orcid":false,"given":"Keigo","family":"Kani","sequence":"first","affiliation":[{"name":"Respiratory Medicine, Rakuwakai Otowa Hospital, Kyoto, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0006-1291-2076","authenticated-orcid":false,"given":"Eri","family":"Shimada","sequence":"additional","affiliation":[{"name":"Diagnostic Pathology, Rakuwakai Otowa Hospital, Kyoto, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0006-9699-4039","authenticated-orcid":false,"given":"Michiko","family":"Tsuchiya","sequence":"additional","affiliation":[{"name":"Respiratory Medicine, Rakuwakai Otowa Hospital, Kyoto, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-3351-2702","authenticated-orcid":false,"given":"Yukio","family":"Nagasaka","sequence":"additional","affiliation":[{"name":"Respiratory Medicine, Rakuwakai Otowa Hospital, Kyoto, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2025,8,11]]},"reference":[{"key":"2025082709585812000_18.8.e266696.1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra1402225"},{"key":"2025082709585812000_18.8.e266696.2","doi-asserted-by":"publisher","DOI":"10.1067\/mjd.2002.120621"},{"key":"2025082709585812000_18.8.e266696.3","doi-asserted-by":"crossref","first-page":"293","DOI":"10.1007\/s12016-020-08822-5","article-title":"Interstitial Lung Disease in Anti-MDA5 Positive Dermatomyositis","volume":"60","author":"Wu","year":"2021","journal-title":"Clin Rev Allergy Immunol"},{"key":"2025082709585812000_18.8.e266696.4","doi-asserted-by":"crossref","first-page":"1535","DOI":"10.1016\/j.chest.2020.04.057","article-title":"Mortality Risk Prediction in Amyopathic Dermatomyositis Associated With Interstitial Lung Disease: The FLAIR Model","volume":"158","author":"Lian","year":"2020","journal-title":"Chest"},{"key":"2025082709585812000_18.8.e266696.5","doi-asserted-by":"crossref","first-page":"359","DOI":"10.1007\/s11926-013-0359-6","article-title":"Interstitial lung disease in inflammatory myopathies: clinical phenotypes and prognosis","volume":"15","author":"Kiely","year":"2013","journal-title":"Curr Rheumatol Rep"},{"key":"2025082709585812000_18.8.e266696.6","doi-asserted-by":"crossref","first-page":"689","DOI":"10.1002\/acr.22728","article-title":"Anti-Melanoma Differentiation-Associated Gene 5 Is Associated With Rapidly Progressive Lung Disease and Poor Survival in US Patients With Amyopathic and Myopathic Dermatomyositis","volume":"68","author":"Moghadam-Kia","year":"2016","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"2025082709585812000_18.8.e266696.7","doi-asserted-by":"crossref","first-page":"488","DOI":"10.1002\/art.41105","article-title":"Multicenter Prospective Study of the Efficacy and Safety of Combined Immunosuppressive Therapy With High-Dose Glucocorticoid, Tacrolimus, and Cyclophosphamide in Interstitial Lung Diseases Accompanied by Anti-Melanoma Differentiation-Associated Gene 5-Positive Dermatomyositis","volume":"72","author":"Tsuji","year":"2020","journal-title":"Arthritis Rheumatol"},{"key":"2025082709585812000_18.8.e266696.8","doi-asserted-by":"crossref","first-page":"3284","DOI":"10.1093\/rheumatology\/keaa123","article-title":"Efficacy of plasma exchange in anti-MDA5-positive dermatomyositis with interstitial lung disease under combined immunosuppressive treatment","volume":"59","author":"Shirakashi","year":"2020","journal-title":"Rheumatology (Oxford)"},{"key":"2025082709585812000_18.8.e266696.9","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMc1900045"},{"key":"2025082709585812000_18.8.e266696.10","doi-asserted-by":"publisher","DOI":"10.1186\/s13054-015-1018-2"},{"key":"2025082709585812000_18.8.e266696.11","doi-asserted-by":"crossref","first-page":"1405","DOI":"10.1007\/s12630-021-02008-z","article-title":"Canadian Airway Focus Group updated consensus-based recommendations for management of the difficult airway: part 2. 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