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病例报告:一例婴儿支气管迪厄拉富瓦病及文献综述

Case Report: A Case of Infant Bronchial Dieulafoy's Disease and Article Review.

作者信息

Chen Yang, Mao Yiting, Cheng Xingfeng, Xiong Ruihua, Lan Ying, Chen Feng, Zhang Furong, Liu Jun, Wu Yanting

机构信息

Wuhan Children's Hospital (Wuhan Maternal and Child Healthcare Hospital), Tongji Medical College, Huazhong University of Science & Technology, Wuhan, China.

The International Peace Maternity and Child Health Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China.

出版信息

Front Pediatr. 2021 Sep 15;9:674509. doi: 10.3389/fped.2021.674509. eCollection 2021.

Abstract

Bronchial Dieulafoy's disease (BDD), characterized by constant diameter arterial malformation, is rare, especially among infants. The pathogenesis and clinical features of pediatric patients are unknown. Misdiagnosis and biopsy operations may lead to potential massive hemorrhage, which endangers the patient's life. Here, we present a case of a 9-month-old boy who was diagnosed with BDD with massive hemoptysis. The boy was cured by embolization of the bronchial artery and was in good health at the 1-year follow-up. In addition, we searched PubMed, Google Scholar, and Web of Science databases using keyword "Bronchial Dieulafoy's Disease (BDD)" and found six additional cases of pediatric BDD. It is still insufficient to draw a conclusion about the origin of the disease. Bronchial angiography and endobronchial ultrasonography are considered promising methods to diagnose Dieulafoy's disease of the bronchus. Bronchoscopy with transbronchial biopsy should not be deployed due to the high risk of fatal hemorrhage. Explicit clinical case reports of BDD are needed to enhance the understanding of this rare disease.

摘要

支气管迪厄拉富瓦病(BDD)以动脉直径恒定的畸形为特征,较为罕见,在婴儿中尤为如此。儿科患者的发病机制和临床特征尚不清楚。误诊和活检操作可能导致潜在的大出血,危及患者生命。在此,我们报告一例9个月大的男孩,他被诊断为BDD并伴有大量咯血。该男孩通过支气管动脉栓塞治愈,在1年的随访中身体健康。此外,我们使用关键词“支气管迪厄拉富瓦病(BDD)”在PubMed、谷歌学术和科学网数据库中进行检索,又发现了6例儿科BDD病例。关于该疾病的起源仍不足以得出结论。支气管血管造影和支气管内超声检查被认为是诊断支气管迪厄拉富瓦病的有前景的方法。由于致命出血风险高,不应进行经支气管活检的支气管镜检查。需要明确的BDD临床病例报告来加强对这种罕见疾病的认识。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2b6e/8480259/b603b62b11e0/fped-09-674509-g0001.jpg

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