Treatment of bronchobiliary fistula: a 13-year experience

Treatment of bronchobiliary fistula: a 13-year experience | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Research Article Treatment of bronchobiliary fistula: a 13-year experience Xi Yu, Yuan Ding, Yi Zhang, Bin Chen, Yun-Peng Hua, Shao-Qiang Li, and 2 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-7418808/v1 This work is licensed under a CC BY 4.0 License Status: Published Journal Publication published 07 Dec, 2025 Read the published version in BMC Surgery → Version 1 posted 12 You are reading this latest preprint version Abstract Background Bronchobiliary fistula (BBF) is a rare but fatal disease. Only a limited number of cases have been reported, resulting in a lack of consensus on appropriate treatment strategies. Methods We retrospectively analyzed the clinical data of 17 patients with BBF between 2012 and 2025, including the main symptoms, diagnosis, treatment and prognosis. Results All 17 patients had cough and pathognomonic biliptysis. Sputum examination revealed bile components in all samples from 3 patients, and fiber bronchoscopy identified yellow-green bilious sputum in 6 patients. Computed tomography (CT)/ magnetic resonance imaging (MRI) demonstrated a connection between the bile duct and the bronchial tree in 9 patients. Cholangiography showed that the contrast agent entered into the bronchi through the sinus tract in 10 patients. Surgical intervention was performed on 6 patients, while 11 underwent minimally invasive treatments, including percutaneous transhepatic cholangial drainage (PTCD), nasobiliary duct, and bile duct stent. During a median 3 years follow-up, 9 of 17 patients survived. Mortality causes included: malignancy progression (n = 3), postoperative complications (pneumothorax/respiratory failure n = 1; hemorrhagic shock/ disseminated intravascular coagulation n = 1), septic shock post-transplant (n = 1), and uncontrolled BBF with septic/hepatic failure (n = 2). Conclusions BBF carries a poor prognosis. Minimally invasive therapies effectively palliate malignant cases, while surgery offers potential cure for select benign disease. Treatment requires individualized, multidisciplinary strategies. Bronchobiliary fistulas (BBF) diagnosis surgery minimally invasive treatment prognosis Figures Figure 1 Figure 2 Figure 3 Figure 4 Introduction Bronchobiliary fistula (BBF) is an abnormal channel between the bronchial tree and the biliary system[ 1 ]. This rare clinical disease can be classified as either congenital or acquired. The etiologies of acquired BBF are diverse, including liver hydatid[ 2 ], thoracic and abdominal trauma[ 3 ], liver malignancies[ 4 ], complications following transcatheter arterial chemoembolization(TACE) or liver radiofrequency ablation[ 5 , 6 ] as well as biliary obstruction[ 7 , 8 ]. BBF classically manifests as refractory cough with bilioptysis, recurrent pulmonary infections, jaundice, and chest/abdominal pain[ 9 ]. Critically, delayed recognition may progress to life-threatening sepsis or respiratory failure[ 10 , 11 ]. Given symptom overlap with pulmonary conditions and frequent misdiagnosis, BBF should be considered in patients with recurrent cough and bitter-tasting sputum, particularly those with hepatobiliary history[ 12 ]. While morphologic differentiation from infectious sputum is challenging, bile component analysis via sputum bilirubin testing offers specificity. Furthermore, fiberoptic bronchoscopy offers direct visualization of both bilious secretions and fistula tracts[ 11 ], with recent evidence supporting bronchoalveolar lavage bilirubin crystals as diagnostic markers[ 13 ]. Imaging examinations such as computed tomography (CT), magnetic resonance imaging (MRI), magnetic resonance cholangiopancreatography (MRCP) and cholangiography also play key roles in evaluating the location and extent of BBF, thus helping assess the disease severity and patient prognosis[ 14 – 16 ]. Historically, BBF has been managed surgically, often involving drainage of subphrenic abscesses and, when necessary, resection of the fistula tract, affected lung tissue, and the primary lesion[ 17 ]. Recently, minimally invasive and endoscopic techniques have broadened treatment options for BBF. For example, endoscopic sphincterotomy and placement of biliary stent has been reported to be an effective treatment method that can relieve biliary obstruction and promote fistula closure[ 15 , 18 , 19 ]. In addition, nasobiliary drainage and percutaneous transhepatic cholangial drainage (PTCD) are used for biliary drainage in patients with poor general conditions and complex conditions[ 18 ]. Other conservative methods, such as abscess drainage, can also enhance the healing of BBF lesions[ 12 ]. Recent years, some studies have shown that bronchial occlusion devices such as glue and coils can effectively eliminate the sinus tract[ 15 , 20 – 22 ]. Despite these advancements, a consensus on the management of BBF remains elusive, and no definitive treatment exists for those complex cases. This study aims to review the characteristics and treatment experiences of 17 patients with BBF in our center from 2012 to 2025, providing insights and guidance for the management of this challenging disease. Materials and Methods Patients This study retrospectively analyzed the clinical data of 17 patients diagnosed with BBF in our center between January 1, 2012 and May 30, 2025. The cohort included 13 males and 4 females, with an average age of 53.5 years. All patients had a history of treatment for primary liver diseases. Four patients had intrahepatic and extrahepatic bile duct stones, 1 patient had hilar cholangiocarcinoma, 1 patient had intrahepatic cholangiocarcinoma (ICC), and 11 patients had hepatocellular carcinoma (HCC). The diagnoses of all primary diseases were confirmed by postoperative pathology examination. For the treatment of the primary disease, 14 patients received surgery, 8 patients received TACE, 6 patients received radiofrequency ablation, and 1 patient received radiotherapy. Table 1 summarizes additional details of the included patients. Results Symptoms and diagnosis All 17 patients had clinical symptoms of cough, pathognomonic biliptysis, and recurrent fever during the disease course. Ten patients had right upper abdominal pain, 3 patients had vomiting, and 4 patients had jaundice. The diagnosis of BBF mainly depended on clinical symptoms, sputum analysis, imaging examination and fiberoptic bronchoscopy. Imaging examinations included CT, MRI, MRCP, ultrasound, contrast-enhanced ultrasound and cholangiography. Sputum analysis was conducted in 3 patients and demonstrated the presence of bile components in all detected samples. Figure 1 showed the characteristic yellow mucinous sputum with a bilious odor observed in patients with BBF. CT showed pneumonia and pleural effusion in all cases. Liver abscess was found in 8 patients, subphrenic abscess in 8 patients, bile duct dilatation in 16 patients, and bile leakage in 12 patients. The imaging examination indicated the presence of BBF in 9 patients, and the representative images were shown in Fig. 2 . Six patients underwent bronchoscopy that confirmed the diagnosis of BBF (Fig. 3 ). Cholangiography was performed in 14 patients, with 10 patients presenting evidences of BBF (Fig. 4 ). Sputum culture was performed in 10 patients, of whom 7 were found to have bacterial infection and 1 was found to have fungal infection. Culture of drainage fluid was performed in 11 patients, of which 6 were found to have bacterial infection and 2 were found to have fungal infection. Treatment and outcome Six patients underwent surgery. Patient 1 underwent fistula resection, diaphragmatic repair, and right hepatic abscess excision with subphrenic drainage placement, achieving symptom resolution and uneventful discharge. Patient 2 received resection of segments VII/VIII, choledocholithotomy, and T-tube drainage, with postoperative symptom relief. Patient 3 received PTCD 1 month before operation to recover the liver function, and then underwent choledocholithotomy and Roux-en-Y choledochojejunostomy, resulting in effective symptom control. All three patients demonstrated resolved thoracic and abdominal infections on 1-year follow-up CT and maintained symptom remission with long-term survival (> 24 months). Patient 4 required two fistula resections within four years. Initial choledocholithotomy with fistula resection failed to resolve symptoms (cough, bilioptysis, fever), necessitating reoperation with right posterior sectionectomy, fistula closure, and common bile duct exploration with stone extraction. Two years postoperatively, readmission for liver abscess precipitated septic shock and pleural effusion, culminating in death from acute obstructive suppurative cholangitis and hepatorenal syndrome. Patient 5 underwent right hemihepatectomy and choledochobronchial fistulectomy. However, this patient developed massive pneumothorax and respiratory failure after surgery, and died of respiratory failure 2 weeks after surgery. Patient 6 underwent partial hepatectomy and partial diaphragm resection and repair. However, the patient developed biliary leakage, septic shock, and hemothorax postoperatively, ultimately succumbing to disseminated intravascular coagulation (DIC), hypovolemic shock, and septic shock two weeks after surgery. Eleven patients received biliary decompression as the prominent treatment for BBF. When the presence of abscess was observed, the interventional drainage methods were adopted. Among these, PTCD was used for biliary drainage in 7 patients, nasobiliary duct was used in 2 patient, and bile duct stent was used for biliary decompression in 3 patients. All patients demonstrated immediate resolution of cough and fever following drainage initiation and antimicrobial therapy. Post-drainage imaging revealed hepatic and pulmonary improvement in 9 patients. However, one patient died four months after replacement of the PTCD tube due to uncontrolled lung infection and liver failure, one patient succumbed to post-transplant septic shock complicated by gastrointestinal hemorrhage six months after biliary stent placement, and 3 patients died from progression of the primary malignancy. Discussion BBF represents a complex condition involving thoracic-abdominal organ injury and diaphragmatic compromise. Integrated analysis of our cohort and prior literature identifies three prerequisites for BBF formation: (1) intra-abdominal necrotic material accumulation, including post-hepatectomy bile leak, inflammatory residue, ablation-related thermal injury, or abscesses[ 23 ]; (2)diaphragmatic injury from radiofrequency ablation, trauma, or surgical resection - particularly when HCC necessitates diaphragm repair[ 5 , 11 ];༈3༉inadequate biliary drainage, including partial or complete obstruction of the bile duct due to tumors, cholangitis, or stones[ 18 , 23 ]. Elevated biliary pressure synergizes with diaphragmatic defects, facilitating bile or pus migration into the thorax via negative pleural pressure. Concomitant pulmonary inflammation and biliary necrosis subsequently erode bronchial walls to establish fistulas[ 7 ]. This triad explains BBF's predominance in hepatic malignancy patients and its characteristically poor prognosis. Despite the grave prognosis associated with BBF, opportunities exist to significantly alleviate symptoms and enhance the quality of life for patients through appropriate management. Beyond cross-sectional imaging, sputum analysis and bronchoscopy provided definitive diagnosis. Moreover, endoscopic retrograde cholangiopancreatography (ERCP) emerged as the primary therapeutic approach, enabling concurrent diagnosis, biliary stenting/nasobiliary drainage, and stone extraction with favorable efficacy. The surgical management of BBF remains a daunting challenge, and a consensus guideline for such procedures is still lacking. BBF involves both the lung and biliary system, making the treatment a comprehensive challenge that requires multidisciplinary cooperation and fine surgical skills. It requires simultaneous management of the lung, liver and biliary tract diseases, as well as repair of the diaphragm defect, including resection of intrahepatic or subphrenic abscess, alleviation or elimination of biliary obstruction, and treatment of the underlying cause of BBF. The traditional surgical strategy is based on the thoracotomy approach, and Hatice Eryigit et al.[ 24 ] reported that thoracotomy was beneficial for patients with persistent fistula and biliary tract patency. However, Tocci[ 17 ] reported that standard thoracotomy was not safe for the treatment of BBF, given the difficulty in controlling the hepatic pedicle, hepatic vein, and inferior vena cava. Although surgery is an effective treatment method, not all patients are suitable because of its high invasiveness and high perioperative mortality. In our center, surgical intervention conferred long-term survival in 3/6 patients but carried significant perioperative mortality. It is necessary to comprehensively consider the patient's condition and choose the most appropriate treatment method. Thus, we advocate for minimally invasive approaches as first-line therapy to achieve triple objectives: effective abscess drainage, prevention of bronchobiliary contamination, and pulmonary infection control. Current strategies comprise two complementary mechanisms: biliary decompression via PTCD, stenting, or nasobiliary drainage and fistula occlusion via embolization or covered stents[ 15 , 22 ]. Endoscopic interventions, particularly sphincterotomy or stenting, serve as effective options by reducing the choledochoduodenal pressure gradient to divert bile flow away from the bronchial tree[ 25 , 26 ]. Nasobiliary drainage or PTCD drainage can also effectively drain the bile and facilitate fistula closure, but there are also risks of electrolyte disturbances and biliary tract infection[ 27 , 28 ]. Recent innovations further expand therapeutic options. For refractory fistulas, advanced techniques including bronchoscopic glue application, coil deployment and bronchial stent demonstrate efficacy. Percutaneous gel embolization employs biocompatible materials for targeted fistula occlusion under imaging guidance, offering low invasiveness and personalized anatomical adaptation[ 3 ]. Similarly, percutaneous endoscopic occlusion enables real-time visual confirmation of defect sealing while minimizing adjacent tissue injury[ 15 , 21 ]. These evolving techniques align with precision medicine paradigms but require optimization of patient selection due to recurrence risks. Future studies should prioritize comparative trials of novel embolization agents and enhanced image-guidance systems to refine procedural accuracy. Our clinical experience supports this paradigm: 10 of 11 patients undergoing minimally invasive decompression achieved significant symptom resolution, with 3 demonstrating long-term survival. Notably, these approaches complement rather than replace surgery. Patients with adequate physiological reserve and resectable disease remain candidates for curative resection[ 5 , 29 ]. Our study has several limitations. First, it is a single-center retrospective analysis, and the case number remains limited although it encompasses the largest sample size reported to date in the extant literature. Second, our cohort primarily consisted of patients with acquired BBF, with limited experience in managing congenital cases. Third, most BBF in this study was secondary to liver malignancy, and the prognosis of patients was influenced by the cancer progression and the complex treatment course. Further multi-center studies are warranted to augment the research methodologies and enhance the reliability of our findings. Conclusion In conclusion, BBF demands a stratified, multidisciplinary management approach. Minimally invasive techniques—encompassing biliary decompression and fistula occlusion—represent the recommended first-line strategy for symptom control and infection mitigation, particularly in malignant or high-risk cases. These approaches align with precision medicine paradigms but require judicious patient selection. Conversely, surgical intervention retains critical importance for anatomically resectable benign disease or complex fistulas in physiologically fit patients. Ultimately, treatment selection must be individualized, integrating endoscopic, radiological, and surgical expertise to address the dual thoracic-biliary pathology. Future efforts should focus on refining patient selection criteria and advancing targeted fistula closure technologies. Abbreviations BBF bronchobiliary fistulas HCC hepatocellular carcinoma TACE transcatheter arterial chemoembolization ICC intrahepatic cholangiocarcinoma CT computerized tomography MRI magnetic resonance imaging PTCD percutaneous transhepatic cholangial drainage MRCP magnetic resonance cholangiopancreatography Declarations Ethics approval and consent to participate: The present study was carried out in accordance with the principles of the Declaration of Helsinki. This retrospective study was approved by the institutional review board of the First Affiliated Hospital of Sun Yat-sen University. Written informed consent was obtained from all patients prior to treatment. Consent for publication: Not applicable. Competing interests: The authors declare that they have no competing interests. Funding Declaration: This work was supported by a grant from the Science and Technology Projects in Guangzhou(2023A04J1809)and Guangdong Basic and Applied Basic Research Foundation (2020A1515010178). The grant was primarily used to cover participant compensation, including research assistant stipends and subject reimbursement. Clinical trial number: not applicable. Author Contribution XY, YD and YZ prepared figures and tables and wrote the manuscript text. BC, YPH and SQL participated in data collection. SLS and ZHD participated in the design of the subject. All authors reviewed the manuscript. Acknowledgments: Not applicable. Authors' information: Department of Hepato-Pancreato-Biliary Surgery, The First Affiliated Hospital of Sun Yat-sen University, Guangzhou, Guangdong, China; Department of Hepatobiliary surgery, the Third Affiliated Hospital, Sun Yat-sen University, No.600 Tian he Road, Guangzhou 510630, China. Data Availability The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request. References Yan CZ, Jia Z, Wan YF, Zhou HS. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-7418808","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":531742977,"identity":"3352a1e4-3cac-468d-8328-3edb1345094d","order_by":0,"name":"Xi Yu","email":"","orcid":"","institution":"The First Affiliated Hospital of Sun Yat-sen University","correspondingAuthor":false,"prefix":"","firstName":"Xi","middleName":"","lastName":"Yu","suffix":""},{"id":531742978,"identity":"7a27a5c6-2100-4a2d-a704-33043364fc44","order_by":1,"name":"Yuan Ding","email":"","orcid":"","institution":"The First Affiliated Hospital of Sun Yat-sen 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01:25:58","extension":"xml","order_by":13,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":99580,"visible":true,"origin":"","legend":"","description":"","filename":"fe9fa914ca774d89b963482eb36d6a191structuring.xml","url":"https://assets-eu.researchsquare.com/files/rs-7418808/v1/24c1da80387062bb9411601d.xml"},{"id":93977923,"identity":"378c31cc-c44b-4871-a802-36a8f655cb4c","added_by":"auto","created_at":"2025-10-21 01:33:58","extension":"html","order_by":14,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":103895,"visible":true,"origin":"","legend":"","description":"","filename":"earlyproof.html","url":"https://assets-eu.researchsquare.com/files/rs-7418808/v1/16263661f17bf53c5c15d72a.html"},{"id":93977415,"identity":"240059ab-5df8-4532-8a37-4f3364d4e07a","added_by":"auto","created_at":"2025-10-21 01:25:58","extension":"jpeg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":26141,"visible":true,"origin":"","legend":"\u003cp\u003eBiliform mucinous sputum coughed up by BBF patients\u003c/p\u003e","description":"","filename":"floatimage1.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-7418808/v1/7e8bc0b6307e1b59f2ddba35.jpeg"},{"id":93977419,"identity":"60d55db8-b198-4e8d-b70f-c7eae0952b7e","added_by":"auto","created_at":"2025-10-21 01:25:58","extension":"jpeg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":757874,"visible":true,"origin":"","legend":"\u003cp\u003eComputed tomography revealed a tunnel between the right bronchus and the biliary tract\u003c/p\u003e","description":"","filename":"floatimage2.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-7418808/v1/e7f8ea7f9c370f89386fe974.jpeg"},{"id":93977919,"identity":"5037da99-36a6-48bf-ab67-2b5f1cd294b6","added_by":"auto","created_at":"2025-10-21 01:33:58","extension":"jpeg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":40233,"visible":true,"origin":"","legend":"\u003cp\u003eBronchoscopic examination of the patient with BBF showed bile-like secretion\u003c/p\u003e","description":"","filename":"groupimage1.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-7418808/v1/4dd2d61c0aae942ae55042d4.jpeg"},{"id":93977922,"identity":"71f773a2-6348-48ae-bc6b-5c83d3dab30a","added_by":"auto","created_at":"2025-10-21 01:33:58","extension":"jpeg","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":60693,"visible":true,"origin":"","legend":"\u003cp\u003eCholangiography in patients with BBF showed communication between bile duct and bronchus\u003c/p\u003e","description":"","filename":"groupimage2.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-7418808/v1/66a5db729b2df867993bf9e8.jpeg"},{"id":97723965,"identity":"5ba5d654-7f84-41b7-84d3-8ce6dcf98b51","added_by":"auto","created_at":"2025-12-08 16:10:17","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":1322565,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-7418808/v1/146486ac-44ba-40be-a07f-325b1c467548.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Treatment of bronchobiliary fistula: a 13-year experience","fulltext":[{"header":"Introduction","content":"\u003cp\u003eBronchobiliary fistula (BBF) is an abnormal channel between the bronchial tree and the biliary system[\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e]. This rare clinical disease can be classified as either congenital or acquired. The etiologies of acquired BBF are diverse, including liver hydatid[\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e], thoracic and abdominal trauma[\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e], liver malignancies[\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e], complications following transcatheter arterial chemoembolization(TACE) or liver radiofrequency ablation[\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e, \u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e] as well as biliary obstruction[\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e, \u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e].\u003c/p\u003e\u003cp\u003eBBF classically manifests as refractory cough with bilioptysis, recurrent pulmonary infections, jaundice, and chest/abdominal pain[\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e]. Critically, delayed recognition may progress to life-threatening sepsis or respiratory failure[\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]. Given symptom overlap with pulmonary conditions and frequent misdiagnosis, BBF should be considered in patients with recurrent cough and bitter-tasting sputum, particularly those with hepatobiliary history[\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e]. While morphologic differentiation from infectious sputum is challenging, bile component analysis via sputum bilirubin testing offers specificity. Furthermore, fiberoptic bronchoscopy offers direct visualization of both bilious secretions and fistula tracts[\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e], with recent evidence supporting bronchoalveolar lavage bilirubin crystals as diagnostic markers[\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e]. Imaging examinations such as computed tomography (CT), magnetic resonance imaging (MRI), magnetic resonance cholangiopancreatography (MRCP) and cholangiography also play key roles in evaluating the location and extent of BBF, thus helping assess the disease severity and patient prognosis[\u003cspan additionalcitationids=\"CR15\" citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e].\u003c/p\u003e\u003cp\u003eHistorically, BBF has been managed surgically, often involving drainage of subphrenic abscesses and, when necessary, resection of the fistula tract, affected lung tissue, and the primary lesion[\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e]. Recently, minimally invasive and endoscopic techniques have broadened treatment options for BBF. For example, endoscopic sphincterotomy and placement of biliary stent has been reported to be an effective treatment method that can relieve biliary obstruction and promote fistula closure[\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e, \u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e, \u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e]. In addition, nasobiliary drainage and percutaneous transhepatic cholangial drainage (PTCD) are used for biliary drainage in patients with poor general conditions and complex conditions[\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e]. Other conservative methods, such as abscess drainage, can also enhance the healing of BBF lesions[\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e]. Recent years, some studies have shown that bronchial occlusion devices such as glue and coils can effectively eliminate the sinus tract[\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e, \u003cspan additionalcitationids=\"CR21\" citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e].\u003c/p\u003e\u003cp\u003eDespite these advancements, a consensus on the management of BBF remains elusive, and no definitive treatment exists for those complex cases. This study aims to review the characteristics and treatment experiences of 17 patients with BBF in our center from 2012 to 2025, providing insights and guidance for the management of this challenging disease.\u003c/p\u003e"},{"header":"Materials and Methods","content":"\u003cdiv id=\"Sec3\" class=\"Section2\"\u003e\u003ch2\u003ePatients\u003c/h2\u003e\u003cp\u003eThis study retrospectively analyzed the clinical data of 17 patients diagnosed with BBF in our center between January 1, 2012 and May 30, 2025. The cohort included 13 males and 4 females, with an average age of 53.5 years. All patients had a history of treatment for primary liver diseases. Four patients had intrahepatic and extrahepatic bile duct stones, 1 patient had hilar cholangiocarcinoma, 1 patient had intrahepatic cholangiocarcinoma (ICC), and 11 patients had hepatocellular carcinoma (HCC). The diagnoses of all primary diseases were confirmed by postoperative pathology examination. For the treatment of the primary disease, 14 patients received surgery, 8 patients received TACE, 6 patients received radiofrequency ablation, and 1 patient received radiotherapy. Table\u0026nbsp;1 summarizes additional details of the included patients.\u003c/p\u003e\u003c/div\u003e"},{"header":"Results","content":"\u003cdiv id=\"Sec5\" class=\"Section2\"\u003e\u003ch2\u003eSymptoms and diagnosis\u003c/h2\u003e\u003cp\u003eAll 17 patients had clinical symptoms of cough, pathognomonic biliptysis, and recurrent fever during the disease course. Ten patients had right upper abdominal pain, 3 patients had vomiting, and 4 patients had jaundice.\u003c/p\u003e\u003cp\u003eThe diagnosis of BBF mainly depended on clinical symptoms, sputum analysis, imaging examination and fiberoptic bronchoscopy. Imaging examinations included CT, MRI, MRCP, ultrasound, contrast-enhanced ultrasound and cholangiography. Sputum analysis was conducted in 3 patients and demonstrated the presence of bile components in all detected samples. Figure\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e showed the characteristic yellow mucinous sputum with a bilious odor observed in patients with BBF. CT showed pneumonia and pleural effusion in all cases. Liver abscess was found in 8 patients, subphrenic abscess in 8 patients, bile duct dilatation in 16 patients, and bile leakage in 12 patients. The imaging examination indicated the presence of BBF in 9 patients, and the representative images were shown in Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e. Six patients underwent bronchoscopy that confirmed the diagnosis of BBF (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e). Cholangiography was performed in 14 patients, with 10 patients presenting evidences of BBF (Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003e).\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003eSputum culture was performed in 10 patients, of whom 7 were found to have bacterial infection and 1 was found to have fungal infection. Culture of drainage fluid was performed in 11 patients, of which 6 were found to have bacterial infection and 2 were found to have fungal infection.\u003c/p\u003e\u003c/div\u003e\n\u003ch3\u003eTreatment and outcome\u003c/h3\u003e\n\u003cp\u003eSix patients underwent surgery. Patient 1 underwent fistula resection, diaphragmatic repair, and right hepatic abscess excision with subphrenic drainage placement, achieving symptom resolution and uneventful discharge. Patient 2 received resection of segments VII/VIII, choledocholithotomy, and T-tube drainage, with postoperative symptom relief. Patient 3 received PTCD 1 month before operation to recover the liver function, and then underwent choledocholithotomy and Roux-en-Y choledochojejunostomy, resulting in effective symptom control. All three patients demonstrated resolved thoracic and abdominal infections on 1-year follow-up CT and maintained symptom remission with long-term survival (\u0026gt;\u0026thinsp;24 months).\u003c/p\u003e\u003cp\u003ePatient 4 required two fistula resections within four years. Initial choledocholithotomy with fistula resection failed to resolve symptoms (cough, bilioptysis, fever), necessitating reoperation with right posterior sectionectomy, fistula closure, and common bile duct exploration with stone extraction. Two years postoperatively, readmission for liver abscess precipitated septic shock and pleural effusion, culminating in death from acute obstructive suppurative cholangitis and hepatorenal syndrome. Patient 5 underwent right hemihepatectomy and choledochobronchial fistulectomy. However, this patient developed massive pneumothorax and respiratory failure after surgery, and died of respiratory failure 2 weeks after surgery. Patient 6 underwent partial hepatectomy and partial diaphragm resection and repair. However, the patient developed biliary leakage, septic shock, and hemothorax postoperatively, ultimately succumbing to disseminated intravascular coagulation (DIC), hypovolemic shock, and septic shock two weeks after surgery.\u003c/p\u003e\u003cp\u003eEleven patients received biliary decompression as the prominent treatment for BBF. When the presence of abscess was observed, the interventional drainage methods were adopted. Among these, PTCD was used for biliary drainage in 7 patients, nasobiliary duct was used in 2 patient, and bile duct stent was used for biliary decompression in 3 patients. All patients demonstrated immediate resolution of cough and fever following drainage initiation and antimicrobial therapy. Post-drainage imaging revealed hepatic and pulmonary improvement in 9 patients. However, one patient died four months after replacement of the PTCD tube due to uncontrolled lung infection and liver failure, one patient succumbed to post-transplant septic shock complicated by gastrointestinal hemorrhage six months after biliary stent placement, and 3 patients died from progression of the primary malignancy.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eBBF represents a complex condition involving thoracic-abdominal organ injury and diaphragmatic compromise. Integrated analysis of our cohort and prior literature identifies three prerequisites for BBF formation: (1) intra-abdominal necrotic material accumulation, including post-hepatectomy bile leak, inflammatory residue, ablation-related thermal injury, or abscesses[\u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e]; (2)diaphragmatic injury from radiofrequency ablation, trauma, or surgical resection - particularly when HCC necessitates diaphragm repair[\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e];༈3༉inadequate biliary drainage, including partial or complete obstruction of the bile duct due to tumors, cholangitis, or stones[\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e, \u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e]. Elevated biliary pressure synergizes with diaphragmatic defects, facilitating bile or pus migration into the thorax via negative pleural pressure. Concomitant pulmonary inflammation and biliary necrosis subsequently erode bronchial walls to establish fistulas[\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e]. This triad explains BBF's predominance in hepatic malignancy patients and its characteristically poor prognosis.\u003c/p\u003e\u003cp\u003eDespite the grave prognosis associated with BBF, opportunities exist to significantly alleviate symptoms and enhance the quality of life for patients through appropriate management. Beyond cross-sectional imaging, sputum analysis and bronchoscopy provided definitive diagnosis. Moreover, endoscopic retrograde cholangiopancreatography (ERCP) emerged as the primary therapeutic approach, enabling concurrent diagnosis, biliary stenting/nasobiliary drainage, and stone extraction with favorable efficacy.\u003c/p\u003e\u003cp\u003eThe surgical management of BBF remains a daunting challenge, and a consensus guideline for such procedures is still lacking. BBF involves both the lung and biliary system, making the treatment a comprehensive challenge that requires multidisciplinary cooperation and fine surgical skills. It requires simultaneous management of the lung, liver and biliary tract diseases, as well as repair of the diaphragm defect, including resection of intrahepatic or subphrenic abscess, alleviation or elimination of biliary obstruction, and treatment of the underlying cause of BBF. The traditional surgical strategy is based on the thoracotomy approach, and Hatice Eryigit et al.[\u003cspan citationid=\"CR24\" class=\"CitationRef\"\u003e24\u003c/span\u003e] reported that thoracotomy was beneficial for patients with persistent fistula and biliary tract patency. However, Tocci[\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e] reported that standard thoracotomy was not safe for the treatment of BBF, given the difficulty in controlling the hepatic pedicle, hepatic vein, and inferior vena cava. Although surgery is an effective treatment method, not all patients are suitable because of its high invasiveness and high perioperative mortality. In our center, surgical intervention conferred long-term survival in 3/6 patients but carried significant perioperative mortality. It is necessary to comprehensively consider the patient's condition and choose the most appropriate treatment method.\u003c/p\u003e\u003cp\u003eThus, we advocate for minimally invasive approaches as first-line therapy to achieve triple objectives: effective abscess drainage, prevention of bronchobiliary contamination, and pulmonary infection control. Current strategies comprise two complementary mechanisms: biliary decompression via PTCD, stenting, or nasobiliary drainage and fistula occlusion via embolization or covered stents[\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e, \u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e]. Endoscopic interventions, particularly sphincterotomy or stenting, serve as effective options by reducing the choledochoduodenal pressure gradient to divert bile flow away from the bronchial tree[\u003cspan citationid=\"CR25\" class=\"CitationRef\"\u003e25\u003c/span\u003e, \u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e]. Nasobiliary drainage or PTCD drainage can also effectively drain the bile and facilitate fistula closure, but there are also risks of electrolyte disturbances and biliary tract infection[\u003cspan citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e, \u003cspan citationid=\"CR28\" class=\"CitationRef\"\u003e28\u003c/span\u003e]. Recent innovations further expand therapeutic options. For refractory fistulas, advanced techniques including bronchoscopic glue application, coil deployment and bronchial stent demonstrate efficacy. Percutaneous gel embolization employs biocompatible materials for targeted fistula occlusion under imaging guidance, offering low invasiveness and personalized anatomical adaptation[\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e]. Similarly, percutaneous endoscopic occlusion enables real-time visual confirmation of defect sealing while minimizing adjacent tissue injury[\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e, \u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e]. These evolving techniques align with precision medicine paradigms but require optimization of patient selection due to recurrence risks. Future studies should prioritize comparative trials of novel embolization agents and enhanced image-guidance systems to refine procedural accuracy. Our clinical experience supports this paradigm: 10 of 11 patients undergoing minimally invasive decompression achieved significant symptom resolution, with 3 demonstrating long-term survival. Notably, these approaches complement rather than replace surgery. Patients with adequate physiological reserve and resectable disease remain candidates for curative resection[\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e, \u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e].\u003c/p\u003e\u003cp\u003eOur study has several limitations. First, it is a single-center retrospective analysis, and the case number remains limited although it encompasses the largest sample size reported to date in the extant literature. Second, our cohort primarily consisted of patients with acquired BBF, with limited experience in managing congenital cases. Third, most BBF in this study was secondary to liver malignancy, and the prognosis of patients was influenced by the cancer progression and the complex treatment course. Further multi-center studies are warranted to augment the research methodologies and enhance the reliability of our findings.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eIn conclusion, BBF demands a stratified, multidisciplinary management approach. Minimally invasive techniques\u0026mdash;encompassing biliary decompression and fistula occlusion\u0026mdash;represent the recommended first-line strategy for symptom control and infection mitigation, particularly in malignant or high-risk cases. These approaches align with precision medicine paradigms but require judicious patient selection. Conversely, surgical intervention retains critical importance for anatomically resectable benign disease or complex fistulas in physiologically fit patients. Ultimately, treatment selection must be individualized, integrating endoscopic, radiological, and surgical expertise to address the dual thoracic-biliary pathology. Future efforts should focus on refining patient selection criteria and advancing targeted fistula closure technologies.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eBBF\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003ebronchobiliary fistulas\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eHCC\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003ehepatocellular carcinoma\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eTACE\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003etranscatheter arterial chemoembolization\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eICC\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003eintrahepatic cholangiocarcinoma\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eCT\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003ecomputerized tomography\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eMRI\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003emagnetic resonance imaging\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003ePTCD\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003epercutaneous transhepatic cholangial drainage\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eMRCP\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003emagnetic resonance cholangiopancreatography\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate:\u003c/strong\u003e\u003cp\u003eThe present study was carried out in accordance with the principles of the Declaration of Helsinki. This retrospective study was approved by the institutional review board of the First Affiliated Hospital of Sun Yat-sen University. Written informed consent was obtained from all patients prior to treatment.\u003c/p\u003e\u003c/p\u003e\u003cp\u003e\u003cstrong\u003eConsent for publication:\u003c/strong\u003e\u003cp\u003eNot applicable.\u003c/p\u003e\u003c/p\u003e\u003cp\u003e\u003cstrong\u003eCompeting interests:\u003c/strong\u003e\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e\u003c/p\u003e\u003ch2\u003eFunding\u003c/h2\u003e\u003cp\u003eDeclaration: This work was supported by a grant from the Science and Technology Projects in Guangzhou(2023A04J1809)and Guangdong Basic and Applied Basic Research Foundation (2020A1515010178). The grant was primarily used to cover participant compensation, including research assistant stipends and subject reimbursement.\u003c/p\u003e\u003cp\u003eClinical trial number: not applicable.\u003c/p\u003e\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eXY, YD and YZ prepared figures and tables and wrote the manuscript text. BC, YPH and SQL participated in data collection. SLS and ZHD participated in the design of the subject. All authors reviewed the manuscript.\u003c/p\u003e\u003ch2\u003eAcknowledgments:\u003c/h2\u003e\u003cp\u003eNot applicable.\u003c/p\u003e\u003cp\u003eAuthors' information: Department of Hepato-Pancreato-Biliary Surgery, The First Affiliated Hospital of Sun Yat-sen University, Guangzhou, Guangdong, China; Department of Hepatobiliary surgery, the Third Affiliated Hospital, Sun Yat-sen University, No.600 Tian he Road, Guangzhou 510630, China.\u003c/p\u003e\u003ch2\u003eData Availability\u003c/h2\u003e\u003cp\u003eThe datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eYan CZ, Jia Z, Wan YF, Zhou HS. Late onset of biliobronchial fistula - a serious complication of hemihepatectomy for atrophic liver with hepatolithiasis: a case report and review of the literature. J Cardiothorac Surg. 2024;19(1):660.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eCan MF, Peker Y. Management of bronchobiliary fistulas caused by liver hydatid disease. Liver Int. 2008;28(7):1042. author reply 1043.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eMukkada RJ, Antony R, Francis JV, Chettupuzha AP, Augustine P, Venugopal B, Koshy A. Bronchobiliary fistula treated successfully with endoscopic microcoils and glue. Ann Thorac Surg. 2014;98(2):e33\u0026ndash;34.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eChen YX, Deng ZH, Zhao H, Zhou BY, Guo JJ, Zeng G, Qian JX. Bronchobiliary fistula in patient with liver cancer. Hepatobiliary Pancreat Dis Int 2023.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eHuang ZM, Zuo MX, Gu YK, Lai CX, Pan QX, Yi XC, Zhang TQ, Huang JH. Bronchobiliary fistula after ablation of hepatocellular carcinoma adjacent to the diaphragm: Case report and literature review. Thorac Cancer. 2020;11(5):1233\u0026ndash;8.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eSliwinski S, Sammons MK, Koca F, El Youzouri H, Vogl T, Bechstein W. Broncho biliary fistula following interventional radiology for hepatic metastases. Z Gastroenterol. 2024;62(8):1211\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003ePinna AD, Marongiu L, Cadoni S, Luridiana E, Nardello O, Pinna DC. Thoracic extension of hydatid cysts of the liver. Surg Gynecol Obstet. 1990;170(3):233\u0026ndash;8.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003ePokharel P, Panah S, Dabek RJ, Schwarzova K, Araim F, Gupta A. Acquired Bronchobiliary Fistula in a Young Adult Patient With Sepsis: A Case Report. Cureus. 2024;16(1):e53110.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eTrieu M, Weihe EK, Sunwoo BY. Bilioptysis Caused by Bronchobiliary Fistula. Am J Respir Crit Care Med. 2023;208(8):896\u0026ndash;7.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eLee S, Lee JH, Kim HB, Lee IJ. Percutaneous Bronchial Embolization to Treat Intractable Bronchobiliary Fistula. Cardiovasc Intervent Radiol. 2019;42(5):784\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eLiao GQ, Wang H, Zhu GY, Zhu KB, Lv FX, Tai S. Management of acquired bronchobiliary fistula: A systematic literature review of 68 cases published in 30 years. World J Gastroenterol. 2011;17(33):3842\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003ePoullis M, Poullis A. Biliptysis caused by a bronchobiliary fistula. J Thorac Cardiovasc Surg. 1999;118(5):971\u0026ndash;2.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eZhang N, Chen Y, Tang L, Zhou D, Hou T. Diagnosis of bronchobiliary fistula by bilirubin crystallization in the alveolar lavage fluid: case reports and literature review. Ann Palliat Med. 2021;10(6):7121\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eKarabulut N, Cakmak V, Kiter G. Confident diagnosis of bronchobiliary fistula using contrast-enhanced magnetic resonance cholangiography. Korean J Radiol. 2010;11(4):493\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eLv Y, Hu B, Tang S, Zhang Y. Successful treatment of bronchobiliary fistula by histoacryl embolization under ERCP guidance without fluoroscopic guidance. Gastrointest Endosc. 2024;99(3):464\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eDamle N, Sahoo M, Bal C, Tripathi M, Chakraborty P, Arora S, Malapure S, Gupta S, Kumar P. Diagnosis of Bronchobiliary Fistula-Utility of 99 m Tc-Mebrofenin Scan and SPECT/CT. Nucl Med Mol Imaging. 2013;47(2):141\u0026ndash;2.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eTocchi A, Mazzoni G, Miccini M, Drumo A, Cassini D, Colace L, Tagliacozzo S. Treatment of hydatid bronchobiliary fistulas: 30 years of experience. Liver Int. 2007;27(2):209\u0026ndash;14.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eSuzuki E, Fujita Y, Matsuhashi N. Case of bile duct coil embolization for broncho-biliary fistula after radiofrequency ablation (with video). Dig Endosc. 2021;33(4):e79\u0026ndash;80.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eYe L, Hu Y, Qian H, Yu J. Treating microwave ablation-complicated bronchobiliary fistula using endoscopic retrograde cholangiopancreatography. Asian J Surg. 2023;46(12):5946\u0026ndash;7.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eSakai T, Honda T, Matsutani N, Kawamura M. Successful Radiograph-guided Bronchial Occlusion With Silicon Spigots for Bronchobiliary Fistula. Ann Thorac Surg. 2022;114(3):e193\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eShang Y, Bai C, Yao XP, Huang Y, Zhao LJ, Li Q. Transnasal flexible bronchoscopic implantation of a nickel titanium (NiTi) bronchial occlusive device for a bronchobiliary fistula. Endoscopy. 2010;42(Suppl 2):E225\u0026ndash;226.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eAl-Asadi Z, Korona MV, Deipolyi AR. Novel Application of a Shear-Thinning Conformable Embolic Gel for Occlusion of a Bronchobiliary Fistula. Cardiovasc Intervent Radiol. 2025;48(2):277\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eSchuld J, Justinger C, Wagner M, Bohle RM, Kollmar O, Schilling MK, Richter S. Bronchobiliary fistula: a rare complication of hepatic endometriosis. Fertil Steril. 2011;95(2):e804815\u0026ndash;808.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eEryigit H, Oztas S, Urek S, Olgac G, Kurutepe M, Kutlu CA. Management of acquired bronchobiliary fistula: 3 case reports and a literature review. J Cardiothorac Surg. 2007;2:52.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eSingh P, Kumar S, Chandra A. Persistent bronchobiliary fistula managed by endoscopic biliary stenting. Hepatobiliary Surg Nutr. 2017;6(4):290\u0026ndash;1.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eCorso RM, Casadei A, Piraccini E, Agnoletti V, Ricci E, Gambale G. Bronchobiliary fistula: a rare cause of acute dyspnea in emergency department. Eur J Emerg Med. 2012;19(3):203\u0026ndash;4.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eYoon DH, Shim JH, Lee WJ, Kim PN, Shin JH, Kim KM. Percutaneous management of a bronchobiliary fistula after radiofrequency ablation in a patient with hepatocellular carcinoma. Korean J Radiol. 2009;10(4):411\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003ePartrinou V, Dougenis D, Kritikos N, Polydorou A, Vagianos C. Treatment of postoperative bronchobiliary fistula by nasobiliary drainage. Surg Endosc. 2001;15(7):758.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eHermi A, Saadi A, Mokadem S, Boussaffa H, Zaghbib S, Haroun A, Bouzouita A, Derouiche A, Chakroun M, Ben Slama MR. Retrovesical hydatid cyst: an unusual location of hydatid disease about a case series. Ann Med Surg (Lond). 2023;85(4):722\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bmc-surgery","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"bsur","sideBox":"Learn more about [BMC Surgery](http://bmcsurg.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/bsur/default.aspx","title":"BMC Surgery","twitterHandle":"@BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Bronchobiliary fistulas (BBF), diagnosis, surgery, minimally invasive treatment, prognosis","lastPublishedDoi":"10.21203/rs.3.rs-7418808/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-7418808/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eBackground\u003c/h2\u003e\u003cp\u003eBronchobiliary fistula (BBF) is a rare but fatal disease. Only a limited number of cases have been reported, resulting in a lack of consensus on appropriate treatment strategies.\u003c/p\u003e\u003ch2\u003eMethods\u003c/h2\u003e\u003cp\u003eWe retrospectively analyzed the clinical data of 17 patients with BBF between 2012 and 2025, including the main symptoms, diagnosis, treatment and prognosis.\u003c/p\u003e\u003ch2\u003eResults\u003c/h2\u003e\u003cp\u003eAll 17 patients had cough and pathognomonic biliptysis. Sputum examination revealed bile components in all samples from 3 patients, and fiber bronchoscopy identified yellow-green bilious sputum in 6 patients. Computed tomography (CT)/ magnetic resonance imaging (MRI) demonstrated a connection between the bile duct and the bronchial tree in 9 patients. Cholangiography showed that the contrast agent entered into the bronchi through the sinus tract in 10 patients. Surgical intervention was performed on 6 patients, while 11 underwent minimally invasive treatments, including percutaneous transhepatic cholangial drainage (PTCD), nasobiliary duct, and bile duct stent. During a median 3 years follow-up, 9 of 17 patients survived. Mortality causes included: malignancy progression (n\u0026thinsp;=\u0026thinsp;3), postoperative complications (pneumothorax/respiratory failure n\u0026thinsp;=\u0026thinsp;1; hemorrhagic shock/ disseminated intravascular coagulation n\u0026thinsp;=\u0026thinsp;1), septic shock post-transplant (n\u0026thinsp;=\u0026thinsp;1), and uncontrolled BBF with septic/hepatic failure (n\u0026thinsp;=\u0026thinsp;2).\u003c/p\u003e\u003ch2\u003eConclusions\u003c/h2\u003e\u003cp\u003eBBF carries a poor prognosis. Minimally invasive therapies effectively palliate malignant cases, while surgery offers potential cure for select benign disease. Treatment requires individualized, multidisciplinary strategies.\u003c/p\u003e","manuscriptTitle":"Treatment of bronchobiliary fistula: a 13-year experience","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-10-21 01:25:53","doi":"10.21203/rs.3.rs-7418808/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2025-10-24T17:41:53+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-10-19T01:02:43+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-10-14T01:41:07+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"268775835911033508786072105548284234856","date":"2025-10-10T06:58:21+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-10-07T23:06:53+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"36596303184615947363828812482848623932","date":"2025-10-07T22:34:16+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"208379827415386642889522647935456544938","date":"2025-10-07T11:32:11+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-10-07T11:02:59+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2025-10-01T13:50:30+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2025-09-05T07:53:04+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2025-09-04T07:28:11+00:00","index":"","fulltext":""},{"type":"submitted","content":"BMC Surgery","date":"2025-09-04T07:24:46+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-surgery","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"bsur","sideBox":"Learn more about [BMC Surgery](http://bmcsurg.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/bsur/default.aspx","title":"BMC Surgery","twitterHandle":"@BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"aeb7a86b-dc9a-48a6-837e-e8bdc2f55252","owner":[],"postedDate":"October 21st, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"published-in-journal","subjectAreas":[],"tags":[],"updatedAt":"2025-12-08T16:04:23+00:00","versionOfRecord":{"articleIdentity":"rs-7418808","link":"https://doi.org/10.1186/s12893-025-03366-x","journal":{"identity":"bmc-surgery","isVorOnly":false,"title":"BMC Surgery"},"publishedOn":"2025-12-07 15:58:29","publishedOnDateReadable":"December 7th, 2025"},"versionCreatedAt":"2025-10-21 01:25:53","video":"","vorDoi":"10.1186/s12893-025-03366-x","vorDoiUrl":"https://doi.org/10.1186/s12893-025-03366-x","workflowStages":[]},"version":"v1","identity":"rs-7418808","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-7418808","identity":"rs-7418808","version":["v1"]},"buildId":"XKTyCvWXoU3ODBz1xrDgd","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}