Late onset of biliobronchial fistula — a serious complication of hemihepatectomy for atrophic liver with hepatolithiasis: a case report and review of the literature

Abstract

Background

Acquired bronchobiliary fistula (BBF) is a rare but life-threatening complication that can occur as a result of oncological processes, inflammatory reactions, parasite infections, thoracoabdominal trauma, or invasive procedures associated with iatrogenic injury. However, the potential etiology of BBF caused by instrumental issues when using ultrasonic scalpels resulting in diathermy burn and its post-burn effects has never been reported.

Case presentation

Herein, we present a case of a 65-year-old woman who developed BBF one month after hepatectomy and presented with refractory irritating cough accompanied by yellow bitter sputum. The diagnosis was confirmed through detection of bile components in the sputum, fiberoptic bronchoscopy examination, and endoscopic retrograde cholangio-pancreatography (ERCP). Unfortunately, both endobronchial blocker and endoscopic nasobiliary drainage (ENBD) failed to address the condition until surgical removal of the involved subdiaphragmatic fistula followed by pedicled greater omentum tamping were performed. After closure of the fistula during a 5-month follow-up period, she did not experience any further episodes of biloptysis.

Conclusions

For diathermy burn associated BBF, non-surgical management proves difficult due to inadequate or ineffective biliary drainage, so that early surgical intervention should be considered as an effective approach. Moreover, the adhesion between the atrophied liver and the diaphragm may pose a significant risk factor for diathermy burn, necessitating heightened vigilance.

Introduction

Bronchobiliary fistula (BBF) is a rare, but life-threatening complication secondary to congenital or acquired factor [1, 2].The etiology of acquired BBF includes oncological processes, inflammatory reactions, lithiasis, postoperative biliary stricture, parasite infection, thoracoabdominal trauma, invasive procedures associated iatrogenic injury, etc [3,4,5]. Among them, though uncommon, sequelae of surgical device-derived diathermy burn is increasing. To date, no cases of BBF triggered by undesirable diathermy burn during surgery have been reported, and the treatment remains controversial due to the difficulty in deciding between non-surgical management and surgical intervention [5, 6]. In addition, the specific surgical approach is also a matter of discussion. Herein, we report a case of patient with postoperative BBF most likely triggered the diathermy burn and its post-burn effects from the ultrasonic scalpel that separated the adhesion between the atrophied right liver and the diaphragm, aiming to summarize its clinical characteristics, diagnostic methods, and therapeutic experience.

Case presentation

The patient, a 65-year-old woman with a 2-month history of type 2 diabetes and well-managed glycemic control, presented with a body mass index of 26.6. She experiencd a cough that had been irritating for the past month, producing yellow and bitter sputum. She also experienced occasional sensations of nausea and dyspnea. She denied jaundice, abdominal pain, and recent cold symptoms. One month prior to the onset of her cough attacks, she underwent a right hemihepatectomy for hepatolithiasis with atrophy of the right liver. Preoperative computed tomography (CT) revealed close adherence of the atrophied right liver to the diaphragm, which was successfully separated intraoperatively using an ultrasonic scalpel (Fig. 1). The patient was discharged approximately 1 month post-surgery without experiencing any discomfort at that time. Physical examination revealed tachycardia and tachypnea during coughing episodes, as well as reduced breath sounds in the lower right lung. Apart from mildly increased C-reactive protein level, no other abnormal results were found on conventional laboratory testing. Thoracoabdominal CT revealed pleural effusion and partial pulmonary atelectasis in the lower right lung, as well as pneumobilia in liver, suggesting a potential potential BBF (Figs. 2 and 3). Biochemical analysis confirmed BBF diagnosis by detecting bilirubin components in normal saline-diluted sputum samples, however no bacteria were isolated from these samples. Fiberoptic bronchoscopy observed bile outflowing from the suspicious bronchus but failed to identify the bile outlet necessary for endobronchial blocker placement. Endoscopic retrograde cholangio-pancreatography (ERCP) exploring upper right hepatic duct demonstrated that the contrast medium ascended into the affected lung through the diaphragm (Fig. 4). Subsequently, endoscopic nasobiliary drainage (ENBD) was implemented for selected biliary drainage following the outflow tract. Additionally, ultrasound-guided percutaneous drainage of the right subdiaphragmatic space was also performed, but nothing was drawn out. Sadly, the patient showed no improvement after a 1-month wait-and-see approach, as evidenced by the progressive pulmonary lesion observed on reexamination of the chest CT scan, which revealed an enlarged pleural effusion and more pronounced pulmonary atelectasis. Therefore, she underwent surgery. Intraperitoneal exploration revealed that there was a close adhesion of 1 cm diameter connecting the left liver cross-section and the diaphragm, and the surrounding tissue was corroded and the structure was disorganized, suggesting that the fistula was located here. The adhesion between the liver cross-section and the diaphragm was separated by hemostat, and then the liver cross-section and diaphragm were sutured respectively. As a result, the fistula was successfully removed, while subphrenic inflammation were eliminated without any other abnormalities in the diaphragm. Finally, the pedicled greater omentum with vessels was dissected and tamped under the diaphragm. Following this surgery, her bilioptysis promptly resolved postoperatively, while the cough persisted transiently and gradually improved. Consequently, the patient was discharged 6 weeks post-surgery upon achieving complete recovery. There were no relapses or complications during a 5-month postoperative follow-up. During her last outpatient review, no abnormal signs were detected by fiberoptic bronchoscopy.

Fig. 1

Red arrow: the atrophied right liver was closely adherent to the diaphragm

Fig. 2

Red arrow: partial pulmonary atelectasis. Yellow arrow: pleural effusion

Fig. 3

Red arrow: pneumobilia in liver

Fig. 4

Red arrow: the approximate location of the fistula in ERCP. (Note: the image of the contrast medium passing through the fistula was not captured due to the small size of the fistula and the brief duration of contrast medium retention.)

Discussion

BBF is an abnormal communication between the hepatobiliary tract and the bronchial tree, which develops when bile is inadequately drained via the natural biliary system, leading to bilioptysis, biliary obstruction, and relevant inflammatory reactions in the subdiaphragmatic space followed by subsequent rupture through the bronchial system [7]. Clinical symptoms include bilioptysis (100%), fever (53%), jaundice (29%), abdominal pain (20%), and respiratory disorder (12%) [2]. Some cases presenting with an irritating cough accompanied by yellow or green sputum are often associated with suspicious pneumonia, which is importantly suggestive of potential BBF [2, 5, 7]. Reviewing literature, a total of 11 cases with BBF after radiofrequency ablation all presented with cough and bilioptysis, and other clinical manifestations included liver abscess and hepatic biloma [6]. Patients who develop BBF following hepatectomy often exhibit infectious inflammation, which is believed to contribute to the pathogenesis of BBF. Additionally, it has been mentioned that surgical injury may also play a role in the development of BBF after hepatectomy; however, possible forms of surgical injury in this process were not discussed. And the diathermy burn has been recognized as a pathogenesis of BBF after ablation, but it has not been considered in the case of BBF after surgery. In the past two decades, the increasing application of ultrasonic scalpels in surgery has led to a rise in the risk of undesirable intraoperative diathermy burn, which is uncommon but becoming more frequent. This burn can result in various post-burn effects and complications such as traumatic aneurysms. In this case, the accurate cause of BBF remained unknown. The atrophic liver in the patient may be attributed to cholestasis or an inflammatory reaction induced by hepatolithiasis and closely adhered to the diaphragm. This close adhesion might predispose to diathermy burns during tissue separation using an ultrasonic scalpel in hemihepatectomy. The patient with bilioptysis did not exhibit fever and showed no significant elevation in inflammatory index except for a slightly elevated CRP level. The sputum sample tested negative for bacterial presence. Additionally, ultrasound-guided percutaneous drainage of the right subdiaphragmatic space yielded no fluid collection, confirming the absence of infective inflammation beneath the diaphragm. Therefore, as observed during the surgical procedure, it was speculated that the diathermy burn, which exhibited pronounced corrosiveness and had delayed impact on surrounding tissue, was considered to be the most likely pathogenesis. It is generally believed that the diagnosis of BBF is highly reliable as long as we possess a comprehensive understanding of BBF and conduct meticulous differential diagnosis to distinguish it from pneumonia and sputum contaminated by digestive fluid. As we all well know, bilirubin is the major component in bile. Therefore, detecting bilirubin in sputum provides the most direct evidence of BBF. Both urinary dipstick tests and biochemistry tests can confirm the presence of bilirubin. Additionally, ERCP with contrast medium could trace the fistula tract, facilitating further assessment of BBF [2]. Fortunately, abdominal magnetic resonance imaging sometimes reveals the fistula tract as a useful diagnostic supplement [6].

After reviewing the English literature published on Medline in the past 40 years, less than 100 cases of acquired BBF were reported, and nearly half of these cases were cured through conservative treatments, including percutaneous drainage of subphrenic abscess, endobronchial blocker by fiberoptic bronchoscopy, and percutaneous transhepatic cholangiodrainage or ENBD. Generally, a watch-and-wait period of 1–2 weeks is sufficient to assess therapeutic effectiveness or ineffectiveness. Prolonged observation does not alter the initial trend or predictable final outcomes. Therefore, for refractory BBF cases where conservative treatments have proven ineffective, more surgeons are opting for surgical management rather than uncertain or ineffective non-surgical approaches [2, 5,6,7]. And thoracoscopic surgery alone or laparoscopic surgery alone can be used as well as a combination of both techniques or traditional open surgery. All these approaches can effectively address the problem, with the combined thoracoabdominal approach being the most reliable but causing more trauma. If the primary lesion is located in the chest cavity, the thoracic approach is recommended; whereas if it is in the abdominal cavity, then the abdominal approach should be considered. Following multidisciplinary discussions, we believed that for this case, opting for an abdominal approach would facilitate intraoperative exploration of the lesion site and avoid unnecessary additional trauma. In our opinion, the key to successful surgery lies in accurately identifying the fistula site and thoroughly removing any subdiaphragmatic foci or cutting off their connection between the bronchial tree and diaphragm. In some cases where it is difficult to separate the involved fistula due to intense adhesion or severe inflammatory reactions surrounding it, local limited en-bloc resection of suspicious foci is also clinically feasible and accessible. And pedicled greater omentum tamping in the subdiaphragmatic space would be favorable for absorbing residual inflammation and closing the fistula. Finally, we propose three principles in the management of BBF as follows: (1) Meticulous assessment through multidisciplinary discussion is necessary before considering therapeutic options; (2) Adopting a step-up approach as a therapeutic strategy, starting with non-invasive methods and progressing to minimally invasive, less invasive, and finally invasive methods; (3) Surgery should be chosen without hesitation when conservative therapies are proven ineffective or uncertain. Additionally, for diathermy burn associated BBF after hepatectomy, we advocate surgery due to a reasonable speculation based on this case that the diathermy burn may have affected the small peripheral bile ducts, resulting in stricture or even occlusion. This can subsequently lead to the late onset of BBF that often does not respond to biliary drainage. And regarding the prevention of BBF during hepatectomy, we believe that the following measures should be focused on: identifying high-risk patients with close adhesion between the liver and diaphragm before surgery; employing energy devices appropriately and performing operations with utmost care; meticulously ensuring hemostasis and inspecting the liver cross-section and diaphragm.

Conclusion

Late onset of biliobronchial fistula after hepatectomy is believed to be associated with burn from ultrasonic scalpel-derived diathermy and postoperative localized biliary stricture, which often does not respond well to conventional conservative treatments. Early surgical removal of the subphrenic fistula and greater omentum may be the optimal therapeutic option for this special sequelae of post-burn effects. In addition, the maintenance of a high level of vigilance is essential in cases where the adhesion between the atrophied liver and diaphragm presents a potential risk for diathermy burn.