Congenital Bronchobiliary Fistula: A Case Report and Literature Review

Bing, Zhen; Chen, Rui; Xing, Pengchao; Ren, Yanling; Hou, Kefeng

doi:10.3389/fped.2021.686827

Cited by 5 publications

(3 citation statements)

References 30 publications

(34 reference statements)

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“…The age of onset and severity of symptoms depend on the diameter of the fistula. Therefore, the symptoms appear at any age from newborn to adults [9,10,29] . Typical clinical feature of CBBF is bilious sputum or bile-stained sputum in tracheal intubation.…”

Section: Searching Resultsmentioning

confidence: 99%

Diagnosis of congenital bronchobiliary fistula in children by bilirubin crystallization in the bronchoalveolar lavage fluid: A case report and literature review

Zhang¹,

Wang²,

Wang³

et al. 2023

Preprint

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Background: Congenital bronchobiliary fistula (CBBF) is a rare disease with abnormal connection between the biliary tract and the bronchus. The misdiagnosis rate of CBBF is high, delayed surgery may induce poor outcome. Case presentation: We reported a girl with CBBF who was confirmed via bronchoscopy and chest computed tomography (CT). The girl was 7-month-old admitted to the hospital with “recurrent pneumonia”. Bilirubin crystallization detection was positive in the bronchoalveolar lavage fluid (BALF). CT images showed the abnormal traffic between the biliary tract and the right main bronchus, the girl was diagnosed with CBBF and recovered after surgical operation. Literature search: A total of 27 cases were collected, 14 (51.8%) cases were diagnosed in the neonatal period, 9 (33.3%) patients were diagnosed in infancy. CBBF was more common in female (19/27, 70.3%). The specific manifestations were bilious sputum (24/27, 88.9%) and bilious vomiting (7/27, 22.2%). Recurrent pneumonia (9/27, 33.3%) was common clinical manifestations. The most abnormal fistula originates different location around the carina was right main bronchus (21/27, 77.8%). There were 23(85.2%) cases recovered after surgical operation and 4(14.8%) cases with severe comorbidities/complications died. Conclusions: CBBF should be suspected in infants with bilious sputum. Bronchoscopy plays crucial role in preoperative evaluation and postoperative follow-up. With early diagnosis and surgery, the prognosis is quite well for CBBF patients without severe biliary malformation and/or severe postoperative complications.

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Section: Searching Resultsmentioning

confidence: 99%

Diagnosis of congenital bronchobiliary fistula in children by bilirubin crystallization in the bronchoalveolar lavage fluid: A case report and literature review

Zhang¹,

Wang²,

Wang³

et al. 2023

Preprint

View full text Add to dashboard Cite

show abstract

“…Therefore, the symptoms appear at any age from newborn to adults [9,10,29]. Typical clinical feature of CBBF is bilious sputum or bile-stained sputum in tracheal intubation.…”

Section: Discussionmentioning

confidence: 99%

Diagnosis of congenital bronchobiliary fistula in children by bilirubin crystallization in the bronchoalveolar lavage fluid: A case report and literature review

Luo

2023

J Clin Images Med Case Rep

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“…As a result, 42 cases were reviewed and data on demographic details, type of fistula, associated anomaly, management, and reported outcomes were collected. The data was abstracted into the evidence table template ( Table 1 [ 1 - 39 ]).…”

Section: Study Identificationmentioning

confidence: 99%

Congenital bilio-bronchial fistula in an adult: a review of literature and video demonstration of laparoscopic fistula tract excision

Murugesan,

Bai,

Pottakkat

et al. 2024

J Minim Invasive Surg

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This article presents a review of the literature on congenital bilio-bronchial fistula (BBF), a rare anomaly characterized by abnormal communication between the bile duct and respiratory tract. Congenital BBF often presents with bilioptysis in early neonates and infants; however, patients with no overt symptoms may occasionally present in adulthood. Our literature search in Medline from 1850 to 2023 revealed 42 reported cases of congenital BBF, primarily managed with thoracotomy and excision of the fistula tract. About one-third of these cases required multiple surgeries due to associated biliary anomalies. The review underscores the importance of diagnostic imaging, including bronchoscopy, in identifying and delineating the extent of the fistula. It also highlights the evolving surgical management, with recent cases showing the efficacy of minimally invasive approaches such as laparoscopy and thoracoscopy. In addition to the literature review, we report a young female patient with a history of recurrent respiratory infections presenting with bilioptysis and extensive left lung damage. Initial management included bronchoscopy-guided glue instillation, left thoracotomy, and pneumonectomy. Following the recurrence of symptoms, the patient was successfully treated with laparoscopic excision of the fistula tract. In recent times, minimally invasive approaches such as laparoscopy and thoracoscopy, with excision of the fistula tract are gaining popularity and have shown good results. We suggest biliary communication being the high-pressure end, tackling it transabdominal may prevent recurrent problems.

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Congenital Bronchobiliary Fistula: A Case Report and Literature Review

Cited by 5 publications

References 30 publications

Diagnosis of congenital bronchobiliary fistula in children by bilirubin crystallization in the bronchoalveolar lavage fluid: A case report and literature review

Diagnosis of congenital bronchobiliary fistula in children by bilirubin crystallization in the bronchoalveolar lavage fluid: A case report and literature review

Diagnosis of congenital bronchobiliary fistula in children by bilirubin crystallization in the bronchoalveolar lavage fluid: A case report and literature review

Congenital bilio-bronchial fistula in an adult: a review of literature and video demonstration of laparoscopic fistula tract excision

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