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病例报告:胆道狭窄后胆支气管瘘

Case report: Biliobronchial fistula after biliary tract stenosis.

作者信息

Batalin Júnior Luís Maurício, Zandoná Mariana Conceição E Silva Seleme, Vargas Thomaz Almeida, de Oliveira Julio Cesar, Chiappetto Juliana Rocha Souza, Oliveira Cassio Vieira, Romeiro Fernando Gomes, Tanni Suzana Erico

机构信息

Pulmonary Division of Internal Medicine, Botucatu School of Medicine-UNESP, São Paulo, Brazil.

Gastroenterology Division of Internal Medicine, Botucatu School of Medicine-UNESP, São Paulo, Brazil.

出版信息

Front Med (Lausanne). 2022 Dec 16;9:1075745. doi: 10.3389/fmed.2022.1075745. eCollection 2022.

DOI:10.3389/fmed.2022.1075745
PMID:36590971
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9800597/
Abstract

Biliobronchial fistula (BBF) is a rare abnormality resulting from congenital or acquired communication between the bile ducts and the bronchial tree. Patients often suffer from chronic cough, dyspnea, and bilioptysis, a pathognomonic symptom of this condition. Conservative methods such as less-invasive procedures are gradually consolidating. Nonetheless, surgery remains the primary treatment, especially in more complex cases. We present the case of a 44-year-old woman with a chronic cough, no verified periods of fever, cyclic jaundice, and episodes of yellowish sputum. She had undergone cholecystectomy in 2018 and had been hospitalized several times since for pneumonia treatment. All consequent investigations for mycobacteriosis were negative. When referred to our hospital, she had cyclic jaundice and parenchymal consolidation in the right lower lobe. Suspected bilioptysis motivated the search for a biliobronchial fistula. Magnetic resonance cholangiography (MRC) confirmed stenosis of the biliary tract and fistulous path, and sputum analysis indicated high bilirubin levels. External biliary bypass was performed as an initial conservative and definitive therapy due to the presence of liver cirrhosis. Although BBF is a rare condition when bilioptysis is suspected, a diagnostic investigation should be initiated. Our case study proposes two criteria for diagnosis: an imaging exam demonstrating the fistulous path and confirmation of bilirubin in the sputum or bronchoalveolar lavage (BAL). When diagnosed, surgical correction should be performed.

摘要

胆支气管瘘(BBF)是一种罕见的异常情况,由胆管与支气管树之间的先天性或后天性连通引起。患者常伴有慢性咳嗽、呼吸困难和咯胆,这是该病症的特征性症状。诸如微创操作等保守方法正逐渐得到巩固。尽管如此,手术仍是主要治疗手段,尤其是在更复杂的病例中。我们报告了一例44岁女性患者,她有慢性咳嗽,无确诊的发热期、周期性黄疸以及淡黄色痰液发作。她于2018年接受了胆囊切除术,此后因肺炎治疗多次住院。所有后续针对分枝杆菌病的检查均为阴性。转诊至我院时,她有周期性黄疸以及右下叶实质性实变。疑似咯胆促使对胆支气管瘘进行排查。磁共振胆管造影(MRC)证实了胆道狭窄和瘘道,痰液分析显示胆红素水平升高。由于存在肝硬化,遂进行了外引流作为初始的保守性和确定性治疗。尽管BBF是一种罕见病症,但当怀疑有咯胆时,应启动诊断性检查。我们的病例研究提出了两条诊断标准:影像学检查显示瘘道以及痰液或支气管肺泡灌洗(BAL)中胆红素得到确认。确诊后,应进行手术矫正。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/9ebdb86f002a/fmed-09-1075745-g0004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/0879265442f4/fmed-09-1075745-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/a8cf901cc689/fmed-09-1075745-g0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/f513643ddb2c/fmed-09-1075745-g0003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/9ebdb86f002a/fmed-09-1075745-g0004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/0879265442f4/fmed-09-1075745-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/a8cf901cc689/fmed-09-1075745-g0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/f513643ddb2c/fmed-09-1075745-g0003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/620c/9800597/9ebdb86f002a/fmed-09-1075745-g0004.jpg

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本文引用的文献

1
Bronchobiliary fistula after stenting of biliary duct as the management of iatrogenic bile duct injury during elective cholecystectomy.胆管支架置入术后发生支气管胆瘘,作为择期胆囊切除术中医源性胆管损伤的处理方式。
Acta Chir Belg. 2023 Oct;123(5):544-549. doi: 10.1080/00015458.2022.2049071. Epub 2022 Mar 11.
2
Congenital Bronchobiliary Fistula with Bright Yellow Serous Sputum.
J Pediatr. 2022 Apr;243:235-236. doi: 10.1016/j.jpeds.2021.12.002. Epub 2021 Dec 7.
3
Diagnosis of bronchobiliary fistula by bilirubin crystallization in the alveolar lavage fluid: case reports and literature review.诊断肺泡灌洗液中胆红素结晶的支气管胆瘘:病例报告和文献复习。
Ann Palliat Med. 2021 Jun;10(6):7121-7125. doi: 10.21037/apm-21-1040.
4
Spontaneous bronchobiliary fistula: Case report.自发性支气管-肝胆管瘘:病例报告。
J Radiol Case Rep. 2020 Oct 31;14(10):10-15. doi: 10.3941/jrcr.v14i10.3743. eCollection 2020 Oct.
5
Congenital bronchobiliary fistula: a case report and literature review.先天性支气管胆管瘘:一例病例报告及文献综述
Respirol Case Rep. 2021 Feb 28;9(4):e00731. doi: 10.1002/rcr2.731. eCollection 2021 Apr.
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Bronchobiliary Fistula.
Intern Emerg Med. 2021 Jun;16(4):1093-1094. doi: 10.1007/s11739-020-02572-0. Epub 2021 Jan 3.
7
Bronchobiliary fistula after ablation of hepatocellular carcinoma adjacent to the diaphragm: Case report and literature review.膈邻近肝癌消融后并发支气管胆瘘:病例报告及文献复习。
Thorac Cancer. 2020 May;11(5):1233-1238. doi: 10.1111/1759-7714.13380. Epub 2020 Mar 9.
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Post-traumatic bronchobiliary fistula.创伤后支气管胆管瘘
BMJ Case Rep. 2019 Apr 5;12(4):e228294. doi: 10.1136/bcr-2018-228294.
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Case in Which Hydatids Were Expectorated, and One of Suppuration in a Hydatid Cyst of the Liver Communicating with the Lungs.咳出包虫病例以及肝包虫囊肿与肺相通伴化脓的病例。
Edinb Med Surg J. 1850 Jul 1;74(184):33-46.
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Biliary Pneumonia due to the Presence of a Bronchobiliary Fistula.因存在支气管胆管瘘导致的胆汁性肺炎。
Intern Med. 2017;56(11):1451-1452. doi: 10.2169/internalmedicine.56.8066. Epub 2017 Jun 1.