Bronchobiliary fistula in fibrolamellar hepatocellular carcinoma with anesthetic challenges during living donor liver transplantation: A case report

Table 1 Timeline of the patient’s clinical course

Time	Clinical event	Intervention	Outcome/note
2020 (5 years prior)	Diagnosis of FL-HCC	Two cycles of nivolumab	Discontinued due to immune-related type 1 DM and thyroiditis
Within first year	Extensive hepatic tumor burden	Ex vivo hepatic tumor resection with Roux-en-Y hepaticojejunostomy	Major alteration of biliary anatomy
Following years	Tumor recurrence	Microwave ablation and radioembolization	Progressive hepatic injury
4 years after diagnosis	Pulmonary metastasis	Left lung wedge resection and stereotactic radiation to both lungs	Radiation exposure to lung parenchyma
Subsequent course	Development of hepatic bilomas, intrahepatic abscesses, polymicrobial cholangitis	Internal–external biliary drainage	Recurrent sepsis and chronic biliary complications
Later period	Development of BBF	Surgical repair with diaphragmatic reconstruction	Recurrent fistulae and persistent bilioptysis
Preoperative period	Drain malfunction and persistent BBF	IR exchange and repositioning of chest tube, biloma drain, biliary drain	Multiple drains present in right hemithorax and RUQ
Preoperative status (2025)	Dyspnea on exertion, bilioptysis, poor nutritional status (BMI: 16)	Evaluation for surgery	Compromised pulmonary and systemic condition
Final stage	Persistent BBF with metastatic FL-HCC	Referral for living donor liver transplantation	Considered as potential curative option

FL-HCC: Fibrolamellar hepatocellular carcinoma; BMI: Body mass index; DM: Diabetes mellitus; BBF: Brochobiliary fistula; RUQ: Right upper quadrant.

Table 2 Ventilation parameters, oxygenation status, and arterial blood gas analysis during the procedure

	Baseline after anesthesia	After resumed TLV	After native liver out	5 minutes after reperfusion	90 minutes after reperfusion
Ventilation	OLV	TLV	TLV ± OLV	TLV	TLV
FiO2 (%)	100	89	67	70	70
SpO2 (%)	94	99	100	100	99
PIP (cmH2O)	24	20	23	19	18
ABP (mmHg)	137/57	114/54	137/62	125/58	108/58
pH	7.33	7.35	7.42	7.36	7.44
PaCO2 (mmHg)	49	43	52	43	44
PaO2 (mmHg)	83	140	220	202	161
HCO3- (mmol/L)	26	24	33	25	29
Base excess/deficit	-0.1	-1.7	7.9	-0.9	4.6
Hb (g/dL)	8.2	7.8	7.2	8.9	10.6
K+ (mmol/L)	3.6	3.5	4.9	3.5	3.4
Lactate (mmol/L)	1.10	1.10	4.60	4.80	4.00

OLV: One-lung ventilation; TLV: Two-lung ventilation; PIP: Peak inspiratory pressure; ABP: Arterial blood pressure; Hb: Hemoglobin.

Table 3 Comparative review of anesthetic strategies in literatures

	Sex/age	Primary disease	Biloptysis	Fistula affected lung	Pre-existing pulmonary disease	Surgery	Surgical position	Anesthesia	Endotracheal tube	Ventilation mode	Anesthetic challenge
1	Male/44	BBF after pancreaticoduodenectomy	Yes	Right	Pneumonia	Resection of fistula and bilobectomy	Left lateral decubitus position	GA	Left-sided DLT	OLV	Initially saturation decreased but gradually increased. Repeated bronchial lavage
2	Female/64	BBF with thrombosed hepatic artery aneurysm	Yes	Right	No	Deceased donor liver retransplantation	Supine position	GA	37-Fr left-sided DLT	OLV with CPAP in the right lung	After skin closure, approximately 500 mL/minute air leak from a suspected residual right bronchopleural fistula was detected during TLV, so OLV was resumed
3	Male/58	BBF with prior hepatic hydatid cyst resection	Yes	Right	No	Thoracotomy and resection of fistula and lobectomy	Left lateral decubitus position	GA	37-Fr left-sided DLT	OLV	-
4	Male/61	Intrahepatic cholangiocarcinoma	Yes	N/A	No	ERCP	N/A	GA	8.0 mm SLT	TLV	A spontaneous bronchobiliary fistula to the right bronchial tree was identified intraoperatively, and the patient was reintubated with a DLT
5	Male/20	BBF with prior exploratory laparotomy for grade 4 liver injury due to a blunt abdominal trauma	Yes	Right	No	ERCP and placement of a biliary stent	Prone and slight head-up position	MAC	N/A	SV	Temporarily hypotensive initially and thereafter remained hemodynamically stable
6	Male/66	Polycystic kidney disease and ESLD	Yes	NA	No	Combined liver-kidney transplantation	Supine position	GA	SLT and using a fogarty catheter	TLV	Identified intraoperative BBF with significant air leak; selective segmental isolation using a fogarty catheter

BBF: Bronchobiliary fistula; ESLD: End-stage liver disease; ERCP: Endoscopic retrograde cholangiopancreatography; GA: General anesthesia; MAC: Monitored anesthesia care; N/A: Not applicable; DLT: Double-lumen tube; SLT: Single-lumen tube; OLV: One-lung ventilation; CPAP: Continuous positive airway pressure; SV: Spontaneous ventilation.