Broncho-biliary fistula caused by a left hydatic cyst

Broncho-biliary fistula caused by a left hydatic cyst | 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 Broncho-biliary fistula caused by a left hydatic cyst Achref SARRAJ, Mohamed Ben KHALIFA, Firas JAOUED, Mossaab GHANNOUCHI, and 2 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-5377552/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 5 You are reading this latest preprint version Abstract Broncho-biliary fistula (BBF) of hydatid origin is a rare complication that occurs due to an abnormal connection between the biliary tract and the bronchial tree affecting three levels: abdominal, diaphragmatic, and thoracic. We report the case of a 34-year-old female patient with a recent history of jaundice. She was evaluated with a thoracoabdominal CT scan due to biliptysis and left basal pneumonia resistant to antibiotics revealing a hydatid cyst in the left lobe of the liver, measuring 60x82 mm, complicated by a type IB BBF according to Mestiri's classification. She underwent surgery via laparotomy alone, allowing control of all lesions. Radiological control one month postoperatively showed regression of the lesions. BBF of hydatid origin is rare, and left-sided localization is even rarer. The treatment is primarily surgical, and the choice of the surgical approach must be well adapted to the lesions at different levels, especially considering the type of BBF. Hydatid cyst left biliobronchial fistula bilioptysis hepato-bronchial disconnection cystobronchial fistula Figures Figure 1 Figure 2 Introduction Hydatid disease, an infection caused by the ingestion of eggs from the dog tapeworm Echinococcus granulosis, is commonly found in areas with poor sanitation and frequent human-animal interaction. The liver is the primary site of involvement (50–90%), particularly affecting the right lobe. The lungs are also frequently involved, and in rare instances, the kidneys, brain, or other regions may be affected [Noomen et al. 2013 ]. As the hydatid cyst matures in the liver, it can give rise to various complications [Noomen et al. 2013 ] . One common complication is the rupture of hydatid cysts into the biliary tract, occurring at a frequency of 17 to 44%. Among these complications, intrathoracic rupture is observed, occurring in 0.6–16% of cases. A broncho biliary fistula (BBF), though severe due to the multiplicity of lesions, is a less frequent complication primarily characterized by the presence of bile in the sputum, a condition known as bilioptysis [Noomen et al. 2013 ]. The primary mechanical factors contributing to the transdiaphragmatic migration of the cyst include positive abdominal and negative intrathoracic pressures, the vacuum-like action of diaphragmatic movements, and the traumatic effect of the cyst on surrounding structures. The main cause of bronchial erosion and subsequent broncho biliary fistula (BBF) formation is the combination of pulmonary inflammation and the necrotizing action of bile [Noomen et al. 2013 ] . While surgery is universally accepted as the optimal treatment for BBF, debates continue regarding the most effective surgical approach and technique. Case A 34-year-old female patient, with no notable pathological history, initially presented with abdominal pain, jaundice, and fever, which were ascribed to Hepatitis A. Upon reflection, it was determined to be a case of angiocholitis that resolved spontaneously. She later required a hospitalization in the pulmonology department for left Basi thoracic pain accompanied by fever, and a productive cough yielding bitter greenish sputum with a left basal pulmonary infiltrate on standard chest radiography. Despite well-conducted antibiotic therapy, the lack of symptomatic improvement and radiological clearance led to a thoracoabdominal CT scan. This revealed a 60 x 82 mm hydatid cyst in segment II of the liver. The cyst communicated with the dilated left intrahepatic bile ducts via a 6mm fistula FIGURE 1 and with the left lung through a diaphragmatic breach. It opened into the ventrobasal segment of the left lower lobe through a large bronchial fistula. This was classified as a type IB broncho biliary fistula according to Mestiri’s classification [Mesteri et al. 1987 ] . Following adequate preparation, she underwent a left subcostal surgical approach. The exploration discovered a 70mm hydatid cyst in segment II that displaced the lesser gastric curvature and communicated with the left hemithorax through a 40mm diaphragmatic breach FIGURE 2. The procedure involved a cysto-diaphragmatic disconnection, followed by resection of the protruding dome after aspirating a biliary fluid from the cyst. This revealed a 6mm cysto-biliary fistula that was sutured after confirming the absence of hydatid material in the non-dilated bile ducts via intraoperative cholangiography. The bronchial fistula and the diaphragmatic breach were successively closed, followed by drainage of the residual cavity, subhepatic space, and left interhepato-diaphragmatic space. Albendazole was administered before and after surgery. The postoperative course was uneventful, and a thoraco-abdominal CT scan at 1 month postoperatively showed regression of lesions with no hydatid recurrence. Discussion Broncho biliary fistula (FBB) is an abnormal communication between the biliary tract and the bronchial tree. It represents a dreaded complication of hepatic hydatid cysts rupturing into the thorax due to the potential bronchopulmonary and hepatobiliary injuries it can cause. Hydatid cysts are localized in the right lobe in 67% of cases, and typically, these broncho biliary fistulas arise from hydatid cysts located in the hepatic dome. However, in our patient, the broncho biliary fistula is due to a hydatid cyst in the left lobe of the liver, rendering it a unique and rare case. [ Noomen et al. 2013 ; Rabiou et al. 2018 ] . The clinical presentation of BPF is predominantly pulmonary, with abdominal symptoms being less frequent. Bilioptysis, the coughing up of bile, is a key clinical feature, affecting 12.5 to 77.8% of patients as per various studies [Rabiou et al. 2018 ] . Other signs are dominated by vomica, which indicates the rupture of the hepatic hydatid cyst into the lung. Hepatobiliary signs such as jaundice are not specific [Rabiou et al. 2018 ] . It’s crucial to highlight that these symptoms can significantly vary among patients, necessitating a thorough clinical evaluation for precise diagnosis and treatment planning. Imaging is invariably crucial, and each imaging modality offers its unique advantages. Findings from chest X Rays can be typical or atypical; frequently, an opacity in the lower lobe or a pleural effusion is observed. An abdominal ultrasound can be beneficial by delineating the liver’s morphology and potential obstructions in the biliary tree. Computed Tomography (CT) scans assist in differentiating the relationships among the cyst, blood vessels, and bile ducts. It also aids in assessing local extension and identifying a BBF and other potential sites of the disease. The information provided by these imaging studies plays a pivotal role in determining the most appropriate therapeutic planning. The goal of treating broncho biliary fistulas is to close the fistula and address its root cause, typically requiring surgical intervention along with appropriate preoperative preparation. This preparation involves controlling the infection with tailored antibiotic therapy, ensuring effective respiratory physiotherapy, and rebalancing hydro electrolytes and caloric intake [Rabiou et al. 2018 ] . The surgical approach primarily targets five key objectives: first, it addresses the treatment of endothoracic lesions. Second, it involves the management of hepatic lesions following hepato-diaphragmatic disconnection, accomplished through either total or partial pericystectomy. This method is favored over hepatic resection as it prevents unnecessary loss of healthy parenchyma while preserving tissue conducive to healing and regeneration. Third, the approach includes the detection and treatment of biliary fistulas. Fourth, it focuses on repairing the diaphragm. Lastly, the approach ensures adequate drainage of the pleural and hepatic cavities when necessary [Rabiou et al. 2018 ] . Surgical access involved either a laparotomy, thoracotomy, or a thoracoabdominal (TA) incision [Rabiou et al. 2018 ] . The selection of the access route and surgical technique should not rely on the surgeon’s preference but should instead be customized to suit the specific requirements determined by the location, nature, and extent of the disease, particularly focusing on the type of broncho biliary fistula (FBB), along with thorough preoperative radio clinical and biological assessments. [ El Hammoumi et al. 2021 ] . ] . A thoracoabdominal (TA) incision offers adequate access to both chest and hepatic lesions simultaneously, with an acceptable morbidity and mortality rate. Its importance lies in providing the necessary wide access. Indeed, the TA incision ensures maximum comfort and safety, particularly when addressing significant lesions in the pulmonary parenchyma that often necessitate controlled pulmonary resections like lobectomy or segmentectomy. However, the left biliobronchial fistula presents its own imperatives. Indeed, the hepato-diaphragmatic disconnection via exclusive left thoracic approach poses several challenges due to the anatomical relationships of the left liver with neighboring organs, which may lead to iatrogenic injuries, particularly to the stomach [Rabiou et al. 2018 ] . In cases of active biliary-bronchial fistulas with significant flow, drying up the fistula requires hepato-bronchial disconnection, a procedure that can only be performed after ensuring the freedom of the bile ducts. Therefore, laparotomy becomes imperative for confirming and guaranteeing this unobstructed flow of the bile ducts [ El Hammoumi et al. 2021 ] . Postoperative complications are diverse, with septic complications being the most prevalent, attributed to the inherently septic nature of biliary-bronchial fistula (BBF) surgery. The postoperative morbidity and mortality rate remains high, ranging between 12.2% and 50% [Rabiou et al. 2018 ] . In our case where the hepatic hydatid cyst is located on the left lobe and complicated by a Type IB biliobronchial fistula according to Mestiri's classification [Mesteri et al. 1987 ] , laparotomy alone enabled the treatment of lesions across all three levels (thoracic, diaphragmatic, and abdominal) in a single surgical procedure. This approach shortened hospitalization duration and costs, with both immediate and long-term postoperative outcomes being satisfactory. Conclusion Broncho biliary fistula is a rare and hazardous complication of hepatic hydatid cysts, predominantly affecting the right lobe of the liver. Left-sided localization is uncommon and poses unique challenges, necessitating precise evaluation and a well-established therapeutic strategy. Despite advancements in therapeutic and surgical techniques, the prognosis of BBF remains guarded, underscoring the importance of prevention, early diagnosis, and treatment of hydatid cysts before complications arise. Statements and Declarations Consent : informed consent for publication of information and images was provided by the patient. The authors declare that no funds, grants, or other support were received during the preparation of this manuscript. The authors have no relevant financial or non-financial interests to disclose. References Noomen F, Mahmoudi A, Fodha Md, Boudokhane M, Hamdi A, Fodha M. Traitement chirurgical des kystes hydatiques du foie [Internet]. Vol. 8, EMC - Techniques chirurgicales - Appareil digestif. Elsevier BV; 2013. p. 1‑18. Mesteri S, Kilani T, Thameur H, Sassi S. Les migrations thoraciques des kystes hydatiques du foie: proposition d’une classification. Lyon Chir 1987;83:12–6. Rabiou S, Lakranbi M, Ouadnouni Y, Benajah D, Smahi M. Localisation gauche d’une fistule biliobronchique : complication exceptionnelle de l’hydatidose hépatique [Internet]. Vol. 74, Revue de Pneumologie Clinique. Elsevier BV; 2018. p. 502‑7. El Hammoumi M, Kabiri EH. Bilio-bronchial and bilio-pleuro-bronchial fistulas of hydatic origin. Kardiochir Torakochirurgia Pol. 2021;18(4):239-246. Cite Share Download PDF Status: Under Review Version 1 posted Reviewers agreed at journal 08 Dec, 2025 Reviewers invited by journal 29 Aug, 2025 Editor invited by journal 13 Jul, 2025 Editor assigned by journal 04 Nov, 2024 First submitted to journal 02 Nov, 2024 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. 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-5377552","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":507227577,"identity":"78a07996-46a2-4806-9014-a03795691f03","order_by":0,"name":"Achref SARRAJ","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA8klEQVRIie3RMYvCMBTA8Vcf5JYnWdulnyEQ6CT0q7QcdJJbHHWwFJyKszfcd/AjVAJ26eEqeIOT0w2dpIMcl9ZzzdVNMP/hQSA/khAAm+0BY0B6joBC5aRHgEKvnKOR8I4k4EOJSlwJCiPx5lciYccStxcR5ee2riOI04yCKc2+fA7IaiOp3l7fV5pkSMGBtifpzRFXRrInicMG4kVHmIrXBVfGi4UtuehTck0m9NMSRPNbWgIRSBdZgsNFH1KNpZNHri8QlfexVNLL/iNlJaGJRiT4Jq2/z8rnL5mZ/OV2c9D+EfQCt5zmnt02m832NP0CDzBDqIif9jEAAAAASUVORK5CYII=","orcid":"https://orcid.org/0000-0003-1636-6034","institution":"university hospital Taher Sfar of Mahdia","correspondingAuthor":true,"prefix":"","firstName":"Achref","middleName":"","lastName":"SARRAJ","suffix":""},{"id":507227578,"identity":"eb5f6914-9068-4118-97ee-240600f9d6f9","order_by":1,"name":"Mohamed Ben KHALIFA","email":"","orcid":"","institution":"university hospital Taher sfar of Mahdia","correspondingAuthor":false,"prefix":"","firstName":"Mohamed","middleName":"Ben","lastName":"KHALIFA","suffix":""},{"id":507227579,"identity":"1ea9c2c5-b6b9-4643-b8d4-aebe40cc48d1","order_by":2,"name":"Firas JAOUED","email":"","orcid":"","institution":"university hopital Taher Sfar of Mahdia","correspondingAuthor":false,"prefix":"","firstName":"Firas","middleName":"","lastName":"JAOUED","suffix":""},{"id":507227580,"identity":"448040fa-e99a-4e54-a606-a54c23e4f9ff","order_by":3,"name":"Mossaab GHANNOUCHI","email":"","orcid":"","institution":"university hospital Taher Sfar of Mahdia","correspondingAuthor":false,"prefix":"","firstName":"Mossaab","middleName":"","lastName":"GHANNOUCHI","suffix":""},{"id":507227581,"identity":"4e459d76-5acc-4b15-b015-2d750413553f","order_by":4,"name":"Mahmoud FODHA","email":"","orcid":"","institution":"university hospital Taher Sfar of Mahdia","correspondingAuthor":false,"prefix":"","firstName":"Mahmoud","middleName":"","lastName":"FODHA","suffix":""},{"id":507227582,"identity":"dc67aad7-ed0a-464c-99a8-a91140d38990","order_by":5,"name":"Moez BOUDOKHANE","email":"","orcid":"","institution":"university hospital Taher Sfar of Mahdia","correspondingAuthor":false,"prefix":"","firstName":"Moez","middleName":"","lastName":"BOUDOKHANE","suffix":""}],"badges":[],"createdAt":"2024-11-02 09:52:13","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-5377552/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-5377552/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":90804946,"identity":"4b3679b4-12d0-45fe-9b11-482bd56175b2","added_by":"auto","created_at":"2025-09-08 10:41:03","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":468636,"visible":true,"origin":"","legend":"\u003cp\u003eSee image above for figure legend.\u003c/p\u003e","description":"","filename":"1.png","url":"https://assets-eu.researchsquare.com/files/rs-5377552/v1/b9804d4b1bea4689ab9b9b95.png"},{"id":90804945,"identity":"5fa743c6-102b-40ce-a073-544e3d730885","added_by":"auto","created_at":"2025-09-08 10:41:02","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":324843,"visible":true,"origin":"","legend":"\u003cp\u003eSee image above for figure legend.\u003c/p\u003e","description":"","filename":"2.png","url":"https://assets-eu.researchsquare.com/files/rs-5377552/v1/b8fd16f5bff0a5cac330ff6e.png"},{"id":90804960,"identity":"9ef008ac-c1e6-49e6-9876-b5e67e793ac4","added_by":"auto","created_at":"2025-09-08 10:41:08","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":1426703,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-5377552/v1/f4d95a72-bb97-4657-8651-e85799759f57.pdf"}],"financialInterests":"","formattedTitle":"\u003cp\u003eBroncho-biliary fistula caused by a left hydatic cyst\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eHydatid disease, an infection caused by the ingestion of eggs from the dog tapeworm Echinococcus granulosis, is commonly found in areas with poor sanitation and frequent human-animal interaction. The liver is the primary site of involvement (50\u0026ndash;90%), particularly affecting the right lobe. The lungs are also frequently involved, and in rare instances, the kidneys, brain, or other regions may be affected [Noomen et al. \u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e2013\u003c/span\u003e\u003cb\u003e].\u003c/b\u003e\u003c/p\u003e\u003cp\u003eAs the hydatid cyst matures in the liver, it can give rise to various complications [Noomen et al. \u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e2013\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e. One common complication is the rupture of hydatid cysts into the biliary tract, occurring at a frequency of 17 to 44%. Among these complications, intrathoracic rupture is observed, occurring in 0.6\u0026ndash;16% of cases. A broncho biliary fistula (BBF), though severe due to the multiplicity of lesions, is a less frequent complication primarily characterized by the presence of bile in the sputum, a condition known as bilioptysis [Noomen et al. \u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e2013\u003c/span\u003e\u003cb\u003e].\u003c/b\u003e\u003c/p\u003e\u003cp\u003eThe primary mechanical factors contributing to the transdiaphragmatic migration of the cyst include positive abdominal and negative intrathoracic pressures, the vacuum-like action of diaphragmatic movements, and the traumatic effect of the cyst on surrounding structures.\u003c/p\u003e\u003cp\u003eThe main cause of bronchial erosion and subsequent broncho biliary fistula (BBF) formation is the combination of pulmonary inflammation and the necrotizing action of bile [Noomen et al. \u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e2013\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eWhile surgery is universally accepted as the optimal treatment for BBF, debates continue regarding the most effective surgical approach and technique.\u003c/p\u003e"},{"header":"Case","content":"\u003cp\u003eA 34-year-old female patient, with no notable pathological history, initially presented with abdominal pain, jaundice, and fever, which were ascribed to Hepatitis A. Upon reflection, it was determined to be a case of angiocholitis that resolved spontaneously.\u003c/p\u003e\u003cp\u003eShe later required a hospitalization in the pulmonology department for left Basi thoracic pain accompanied by fever, and a productive cough yielding bitter greenish sputum with a left basal pulmonary infiltrate on standard chest radiography.\u003c/p\u003e\u003cp\u003eDespite well-conducted antibiotic therapy, the lack of symptomatic improvement and radiological clearance led to a thoracoabdominal CT scan. This revealed a 60 x 82 mm hydatid cyst in segment II of the liver. The cyst communicated with the dilated left intrahepatic bile ducts via a 6mm fistula FIGURE 1 and with the left lung through a diaphragmatic breach. It opened into the ventrobasal segment of the left lower lobe through a large bronchial fistula. This was classified as a type IB broncho biliary fistula according to Mestiri\u0026rsquo;s classification [Mesteri et al. \u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e1987\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eFollowing adequate preparation, she underwent a left subcostal surgical approach. The exploration discovered a 70mm hydatid cyst in segment II that displaced the lesser gastric curvature and communicated with the left hemithorax through a 40mm diaphragmatic breach FIGURE 2.\u003c/p\u003e\u003cp\u003eThe procedure involved a cysto-diaphragmatic disconnection, followed by resection of the protruding dome after aspirating a biliary fluid from the cyst. This revealed a 6mm cysto-biliary fistula that was sutured after confirming the absence of hydatid material in the non-dilated bile ducts via intraoperative cholangiography.\u003c/p\u003e\u003cp\u003eThe bronchial fistula and the diaphragmatic breach were successively closed, followed by drainage of the residual cavity, subhepatic space, and left interhepato-diaphragmatic space.\u003c/p\u003e\u003cp\u003eAlbendazole was administered before and after surgery. The postoperative course was uneventful, and a thoraco-abdominal CT scan at 1 month postoperatively showed regression of lesions with no hydatid recurrence.\u003c/p\u003e\u003cp\u003e\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eBroncho biliary fistula (FBB) is an abnormal communication between the biliary tract and the bronchial tree. It represents a dreaded complication of hepatic hydatid cysts rupturing into the thorax due to the potential bronchopulmonary and hepatobiliary injuries it can cause. Hydatid cysts are localized in the right lobe in 67% of cases, and typically, these broncho biliary fistulas arise from hydatid cysts located in the hepatic dome. However, in our patient, the broncho biliary fistula is due to a hydatid cyst in the left lobe of the liver, rendering it a unique and rare case.\u003cb\u003e[\u003c/b\u003e Noomen et al. \u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e2013\u003c/span\u003e ; Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eThe clinical presentation of BPF is predominantly pulmonary, with abdominal symptoms being less frequent.\u003c/p\u003e\u003cp\u003eBilioptysis, the coughing up of bile, is a key clinical feature, affecting 12.5 to 77.8% of patients as per various studies [Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eOther signs are dominated by vomica, which indicates the rupture of the hepatic hydatid cyst into the lung. Hepatobiliary signs such as jaundice are not specific [Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eIt\u0026rsquo;s crucial to highlight that these symptoms can significantly vary among patients, necessitating a thorough clinical evaluation for precise diagnosis and treatment planning. Imaging is invariably crucial, and each imaging modality offers its unique advantages.\u003c/p\u003e\u003cp\u003eFindings from chest X Rays can be typical or atypical; frequently, an opacity in the lower lobe or a pleural effusion is observed. An abdominal ultrasound can be beneficial by delineating the liver\u0026rsquo;s morphology and potential obstructions in the biliary tree.\u003c/p\u003e\u003cp\u003eComputed Tomography (CT) scans assist in differentiating the relationships among the cyst, blood vessels, and bile ducts. It also aids in assessing local extension and identifying a BBF and other potential sites of the disease.\u003c/p\u003e\u003cp\u003eThe information provided by these imaging studies plays a pivotal role in determining the most appropriate therapeutic planning.\u003c/p\u003e\u003cp\u003eThe goal of treating broncho biliary fistulas is to close the fistula and address its root cause, typically requiring surgical intervention along with appropriate preoperative preparation.\u003c/p\u003e\u003cp\u003eThis preparation involves controlling the infection with tailored antibiotic therapy, ensuring effective respiratory physiotherapy, and rebalancing hydro electrolytes and caloric intake [Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eThe surgical approach primarily targets five key objectives: first, it addresses the treatment of endothoracic lesions. Second, it involves the management of hepatic lesions following hepato-diaphragmatic disconnection, accomplished through either total or partial pericystectomy. This method is favored over hepatic resection as it prevents unnecessary loss of healthy parenchyma while preserving tissue conducive to healing and regeneration. Third, the approach includes the detection and treatment of biliary fistulas. Fourth, it focuses on repairing the diaphragm. Lastly, the approach ensures adequate drainage of the pleural and hepatic cavities when necessary [Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eSurgical access involved either a laparotomy, thoracotomy, or a thoracoabdominal (TA) incision [Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eThe selection of the access route and surgical technique should not rely on the surgeon\u0026rsquo;s preference but should instead be customized to suit the specific requirements determined by the location, nature, and extent of the disease, particularly focusing on the type of broncho biliary fistula (FBB), along with thorough preoperative radio clinical and biological assessments. \u003cb\u003e[\u003c/b\u003eEl Hammoumi et al. 2021\u003cb\u003e]\u003c/b\u003e.\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eA thoracoabdominal (TA) incision offers adequate access to both chest and hepatic lesions simultaneously, with an acceptable morbidity and mortality rate. Its importance lies in providing the necessary wide access. Indeed, the TA incision ensures maximum comfort and safety, particularly when addressing significant lesions in the pulmonary parenchyma that often necessitate controlled pulmonary resections like lobectomy or segmentectomy.\u003c/p\u003e\u003cp\u003eHowever, the left biliobronchial fistula presents its own imperatives. Indeed, the hepato-diaphragmatic disconnection via exclusive left thoracic approach poses several challenges due to the anatomical relationships of the left liver with neighboring organs, which may lead to iatrogenic injuries, particularly to the stomach [Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eIn cases of active biliary-bronchial fistulas with significant flow, drying up the fistula requires hepato-bronchial disconnection, a procedure that can only be performed after ensuring the freedom of the bile ducts. Therefore, laparotomy becomes imperative for confirming and guaranteeing this unobstructed flow of the bile ducts \u003cb\u003e[\u003c/b\u003eEl Hammoumi et al. 2021\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003ePostoperative complications are diverse, with septic complications being the most prevalent, attributed to the inherently septic nature of biliary-bronchial fistula (BBF) surgery.\u003c/p\u003e\u003cp\u003eThe postoperative morbidity and mortality rate remains high, ranging between 12.2% and 50% [Rabiou et al. \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e2018\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e.\u003c/p\u003e\u003cp\u003eIn our case where the hepatic hydatid cyst is located on the left lobe and complicated by a Type IB biliobronchial fistula according to Mestiri's classification [Mesteri et al. \u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e1987\u003c/span\u003e\u003cb\u003e]\u003c/b\u003e, laparotomy alone enabled the treatment of lesions across all three levels (thoracic, diaphragmatic, and abdominal) in a single surgical procedure. This approach shortened hospitalization duration and costs, with both immediate and long-term postoperative outcomes being satisfactory.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eBroncho biliary fistula is a rare and hazardous complication of hepatic hydatid cysts, predominantly affecting the right lobe of the liver. Left-sided localization is uncommon and poses unique challenges, necessitating precise evaluation and a well-established therapeutic strategy. Despite advancements in therapeutic and surgical techniques, the prognosis of BBF remains guarded, underscoring the importance of prevention, early diagnosis, and treatment of hydatid cysts before complications arise.\u003c/p\u003e"},{"header":"Statements and Declarations","content":"\u003cp\u003e\u003cstrong\u003e\u003cem\u003eConsent\u003c/em\u003e\u003c/strong\u003e: informed consent for publication of information and images was provided by the patient.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eThe authors declare that no funds, grants, or other support were received during the preparation of this manuscript.\u003c/em\u003e\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eThe authors have no relevant financial or non-financial interests to disclose.\u003c/em\u003e\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eNoomen F, Mahmoudi A, Fodha Md, Boudokhane M, Hamdi A, Fodha M. Traitement chirurgical des kystes hydatiques du foie [Internet]. Vol. 8, EMC - Techniques chirurgicales - Appareil digestif. Elsevier BV; 2013. p. 1‑18. \u003c/li\u003e\n\u003cli\u003eMesteri S, Kilani T, Thameur H, Sassi S. Les migrations thoraciques des kystes hydatiques du foie: proposition d\u0026rsquo;une classification. Lyon Chir 1987;83:12\u0026ndash;6.\u003c/li\u003e\n\u003cli\u003eRabiou S, Lakranbi M, Ouadnouni Y, Benajah D, Smahi M. Localisation gauche d\u0026rsquo;une fistule biliobronchique : complication exceptionnelle de l\u0026rsquo;hydatidose h\u0026eacute;patique [Internet]. Vol. 74, Revue de Pneumologie Clinique. Elsevier BV; 2018. p. 502‑7.\u003c/li\u003e\n\u003cli\u003eEl Hammoumi M, Kabiri EH. Bilio-bronchial and bilio-pleuro-bronchial fistulas of hydatic origin. Kardiochir Torakochirurgia Pol. 2021;18(4):239-246.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"journal-of-parasitic-diseases","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"jopd","sideBox":"Learn more about [Journal of Parasitic Diseases](https://www.springer.com/journal/12639)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/jopd/default.aspx","title":"Journal of Parasitic Diseases","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false},"keywords":"Hydatid cyst, left biliobronchial fistula, bilioptysis, hepato-bronchial disconnection, cystobronchial fistula","lastPublishedDoi":"10.21203/rs.3.rs-5377552/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-5377552/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"Broncho-biliary fistula (BBF) of hydatid origin is a rare complication that occurs due to an abnormal connection between the biliary tract and the bronchial tree affecting three levels: abdominal, diaphragmatic, and thoracic.\nWe report the case of a 34-year-old female patient with a recent history of jaundice. She was evaluated with a thoracoabdominal CT scan due to biliptysis and left basal pneumonia resistant to antibiotics revealing a hydatid cyst in the left lobe of the liver, measuring 60x82 mm, complicated by a type IB BBF according to Mestiri's classification. She underwent surgery via laparotomy alone, allowing control of all lesions. Radiological control one month postoperatively showed regression of the lesions.\nBBF of hydatid origin is rare, and left-sided localization is even rarer. The treatment is primarily surgical, and the choice of the surgical approach must be well adapted to the lesions at different levels, especially considering the type of BBF.","manuscriptTitle":"Broncho-biliary fistula caused by a left hydatic cyst","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-09-08 10:40:58","doi":"10.21203/rs.3.rs-5377552/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"reviewerAgreed","content":"","date":"2025-12-08T06:56:59+00:00","index":0,"fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-08-29T04:57:09+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"Journal of Parasitic Diseases","date":"2025-07-13T12:45:32+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2024-11-04T07:35:45+00:00","index":"","fulltext":""},{"type":"submitted","content":"Journal of Parasitic Diseases","date":"2024-11-02T05:51:40+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"journal-of-parasitic-diseases","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"jopd","sideBox":"Learn more about [Journal of Parasitic Diseases](https://www.springer.com/journal/12639)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/jopd/default.aspx","title":"Journal of Parasitic Diseases","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false}}],"origin":"","ownerIdentity":"ad9b1197-ee11-457e-a08d-eae36d3a5d65","owner":[],"postedDate":"September 8th, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2025-09-08T10:40:58+00:00","versionOfRecord":[],"versionCreatedAt":"2025-09-08 10:40:58","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-5377552","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-5377552","identity":"rs-5377552","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}