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与原发性骨髓纤维化和芦可替尼治疗相关的继发性肺泡蛋白沉积症：一例尸检病例

Secondary Pulmonary Alveolar Proteinosis Associated with Primary Myelofibrosis and Ruxolitinib Treatment: An Autopsy Case.

作者信息

Sugiura Hiroyuki, Nishimori Hisakazu, Nishii Kazuya, Toji Tomohiro, Fujii Keiko, Fujii Nobuharu, Matsuoka Ken-Ichi, Nakata Koh, Kiura Katsuyuki, Maeda Yoshinobu

机构信息

Department of Hematology, Oncology and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Japan.

Department of Pathology, Okayama University Hospital, Japan.

出版信息

Intern Med. 2020 Aug 15;59(16):2023-2028. doi: 10.2169/internalmedicine.4082-19. Epub 2020 May 23.

DOI:10.2169/internalmedicine.4082-19
PMID:32448830
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7492123/
Abstract

Pulmonary alveolar proteinosis (PAP) is an uncommon lung disorder characterized by the excessive accumulation of surfactant-derived lipoproteins in the pulmonary alveoli and terminal bronchiole. Secondary PAP associated with primary myelofibrosis (PMF) is extremely rare, and to our knowledge, no autopsy case has been reported. We herein report an autopsy case of secondary PAP occurring in a patient with PMF who was treated with the Janus kinase 1/2 inhibitor ruxolitinib. We confirmed a diagnosis of PAP with complications based on the pathological findings at the autopsy. Notably, this case might suggest an association between ruxolitinib treatment and PAP occurrence.

摘要

肺泡蛋白沉积症（PAP）是一种罕见的肺部疾病，其特征是肺泡和终末细支气管中表面活性物质衍生的脂蛋白过度积聚。与原发性骨髓纤维化（PMF）相关的继发性PAP极为罕见，据我们所知，尚无尸检病例报道。我们在此报告一例发生在接受Janus激酶1/2抑制剂鲁索替尼治疗的PMF患者中的继发性PAP尸检病例。我们根据尸检时的病理结果确诊了伴有并发症的PAP。值得注意的是，该病例可能提示鲁索替尼治疗与PAP发生之间存在关联。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d8c/7492123/34cd2809ea28/1349-7235-59-2023-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d8c/7492123/fbe41303fcde/1349-7235-59-2023-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d8c/7492123/d3d86a85cda8/1349-7235-59-2023-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d8c/7492123/34cd2809ea28/1349-7235-59-2023-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d8c/7492123/fbe41303fcde/1349-7235-59-2023-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d8c/7492123/d3d86a85cda8/1349-7235-59-2023-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d8c/7492123/34cd2809ea28/1349-7235-59-2023-g003.jpg

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