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两例支气管迪厄拉富瓦病变伴新的合并症及支气管内消融治疗

Two cases of Dieulafoy lesions of the bronchus with novel comorbid associations and endobronchial ablative management.

作者信息

Sheth Hardik S, Maldonado Fabien, Lentz Robert J

机构信息

D. Y. Patil University School of Medicine, Mumbai, India Division of Allergy, Pulmonary, and Critical Care Medicine Department of Thoracic Surgery, Vanderbilt University School of Medicine, Nashville, TN, USA.

出版信息

Medicine (Baltimore). 2018 Feb;97(8):e9754. doi: 10.1097/MD.0000000000009754.

Abstract

RATIONALE

Dieulafoy lesions are aberrantly large submucosal arteries most frequently associated with gastrointestinal hemorrhage. They are rarely identified in the bronchial submucosa and can cause massive hemoptysis.

PATIENT CONCERNS

We present three episodes of massive hemoptysis in two patients, the first with comorbid Alagille syndrome including multiple cardiac and pulmonary vascular abnormalities and the second with thyroid cancer metastatic to the mediastinum.

DIAGNOSES

All episodes were due to Dieulafoy lesions of the bronchus based on bronchoscopic appearance.

INTERVENTIONS

Bronchoscopic ablation using Nd:YAP laser was attempted both patients.

OUTCOMES

Nd:YAP laser successfully ablated the Dieulafoy lesion in the first case with long-term relief from recurrent hemoptysis. The first episode in the second patient responded to bronchial artery embolization; laser ablation of a different Dieulafoy lesion responsible for the second episode was unsuccessful but additional bronchial artery embolization has provided relief from further episodes.

LESSONS

Bronchoscopic ablation of Dieulafoy lesions of the bronchus can provide durable relief from recurrent symptoms. Clinical and anatomical features should be considered carefully before intervention, which should only be attempted by experienced operators with appropriate ancillary support available.

摘要

原理

Dieulafoy病变是异常粗大的黏膜下动脉,最常与胃肠道出血相关。它们在支气管黏膜下很少见,可导致大量咯血。

患者情况

我们报告了两名患者的三次大量咯血发作,第一名患者合并阿拉吉耶综合征,包括多种心脏和肺血管异常,第二名患者患有甲状腺癌纵隔转移。

诊断

根据支气管镜检查表现,所有发作均归因于支气管的Dieulafoy病变。

干预措施

两名患者均尝试使用钕钇铝石榴石(Nd:YAP)激光进行支气管镜消融。

结果

在第一例中,Nd:YAP激光成功消融了Dieulafoy病变,咯血复发得到长期缓解。第二名患者的首次发作对支气管动脉栓塞有反应;对导致第二次发作的另一个Dieulafoy病变进行激光消融未成功,但额外的支气管动脉栓塞缓解了进一步的发作。

经验教训

支气管镜消融支气管Dieulafoy病变可持久缓解复发症状。干预前应仔细考虑临床和解剖特征,且仅应由有经验的操作者在有适当辅助支持的情况下尝试。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c9c3/5842022/78d38543a7fe/medi-97-e9754-g001.jpg

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