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骨软骨化生型气管支气管病(TPO)合并气管支气管淀粉样变性(TBA)。

Tracheobronchopathia osteochondroplastica (TPO) associated with tracheobronchial amyloidosis (TBA).

作者信息

Song Ju Yeun, Kim Bo-Guen, Zo Sungmin, Park Sujin, Han Joung Ho, Kim Hojoong

机构信息

Division of Pulmonary and Critical Care Medicine, Department of Medicine, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, South Korea.

Department of Pathology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, South Korea.

出版信息

Respir Med Case Rep. 2020 Sep 14;31:101223. doi: 10.1016/j.rmcr.2020.101223. eCollection 2020.

DOI:10.1016/j.rmcr.2020.101223
PMID:32995262
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7502850/
Abstract

Tracheobronchopathia osteochondroplastica (TPO) is an idiopathic disease involving the cartilage rings of the large airway, characterized by submucosal calcified nodules. Localized tracheobronchial amyloidosis (TBA) is another rare disease with localized amyloid deposits in the tracheobronchial tree. The two diseases rarely coincide, and only a few case reports and series have been reported. A patient with dyspnea was referred to our clinic for suspicion of TBA. Chest computed tomography (CT) scan showed marked thickening of the tracheobronchial wall with calcified endobronchial submucosal nodules. The nodules were resected with a Diode Laser under rigid bronchoscopy, and results from the biopsy showed both osteochondroid metaplasia on microscopy in Hematoxylin and Eosin staining and apple-green birefringence on polarized microscopy in Congo red staining. This is a rare case in which microscopic findings of both TPO and TBA were observed on one slide. These findings suggest that localized TBA could be a cause of TPO.

摘要

气管支气管骨软骨化生(TPO)是一种累及大气道软骨环的特发性疾病,其特征为黏膜下钙化结节。局限性气管支气管淀粉样变(TBA)是另一种罕见疾病,在气管支气管树中有局限性淀粉样沉积。这两种疾病很少同时出现,仅有少数病例报告和系列报道。一名因怀疑患有TBA而出现呼吸困难的患者被转诊至我院门诊。胸部计算机断层扫描(CT)显示气管支气管壁明显增厚,伴有支气管内黏膜下钙化结节。在硬质支气管镜下用半导体激光切除结节,活检结果显示苏木精-伊红染色显微镜下可见骨软骨化生,刚果红染色偏振显微镜下可见苹果绿双折射。这是一例罕见病例,在一张切片上观察到了TPO和TBA的微观表现。这些发现提示局限性TBA可能是TPO的一个病因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bac/7502850/ccbc57caf297/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bac/7502850/164585526839/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bac/7502850/03816e30f97d/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bac/7502850/ccbc57caf297/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bac/7502850/164585526839/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bac/7502850/03816e30f97d/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bac/7502850/ccbc57caf297/gr3.jpg

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本文引用的文献

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Unusual combination of tracheobronchopathia osteochondroplastica and AA amyloidosis.气管支气管软骨骨肥厚症与 AA 淀粉样变性的不常见组合。
Yonsei Med J. 2009 Oct 31;50(5):721-4. doi: 10.3349/ymj.2009.50.5.721. Epub 2009 Oct 21.
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Calcium deposition with or without bone formation in the lung.肺部出现伴有或不伴有骨形成的钙沉积。
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