T tube sinus tract duodenal fistula: a rare complication of postoperative choledochoscopy for treating retained intrahepatic stones.

BACKGROUNDS: Postoperative percutaneous choledochoscopy via T tube sinus tract is a common modality for treating retained intrahepatic stones in China. We report a rare complication of postoperative choledochoscopy for treating retained hepatolithiasis: T tube sinus tract duodenal fistula. METHODS: From January 2003 to December 2018, intrahepatic duct stones with or without common bile duct stones were detected in 1031 patients. Fifteen of the 1031 patients with intrahepatic stones developed a T tube sinus tract duodenal fistula that was diagnosed by cholangiography and choledochoscopy. RESULTS: The incidence of T tube sinus tract duodenal fistula in patients with retained intrahepatic stones being treated by postoperative choledochoscopy is 1.45% (15/1031) in this series. The chi-squared test showed that hypoalbuminemia (P = 0.003), long duration of T tube (P = 0.002), and high frequency of procedure (P = 0.008) might be associated with the occurrence of T tube sinus tract duodenal fistula. The logistic regression analysis demonstrated that hypoalbuminemia might be the independent risk factor for this special fistula (P = 0.037). CONCLUSIONS: Hypoalbuminemia, long time placement of T tube in situ and high frequency of procedure are probably the main causes of the T tube sinus tract duodenal fistula. Placement of T tube in correct way and improving nutritional status may be the key points to prevent the formation of T tube sinus tract duodenal fistula.

Management of Hepatolithiasis: Review of the Literature.

PURPOSE OF REVIEW: Hepatolithiasis is a disease characterized by intrahepatic stone formation. In this article, we review the features of this disease and explore the established and emerging treatment modalities. RECENT FINDINGS: Recent reports show an increasing prevalence of hepatolithiasis, likely owed to increased immigration and shifts in the Western diet. New pharmacotherapy options are limited and are often only supportive. Endoscopic intervention still cruxes on removal of impacted stones, though new techniques such as bile duct exploratory lithotomy and lithotripsy continue to advance management. Although hepatectomy of the effected portion of the liver offers definitive therapy, alternative less invasive modalities such as combined endoscopic/interventional radiology modalities have been utilized in select patients. Additionally, liver transplant serves as an option for otherwise incurable hepatolithiasis with coexisting liver dysfunction. Multiple emerging pharmacologic and procedural interventions may provide novel treatment for hepatolithiasis. While definitive therapy remains resection of affected liver segments, these modalities offer hope for less invasive approaches in the future.

Per-operative modified rigid cholangioscopy for removal of intrahepatic stones associated with choledochal cyst in children.

INTRODUCTION: Choledochal cyst (CDC) is often associated with intrahepatic stones (IHSs) in children which necessitate their removal during excision. The endoscopic equipment needed for their clearance such as paediatric flexible cholangioscope and other advanced modalities are not freely available in resource-poor setups. We describe per-operative modified rigid cholangioscopy using rigid paediatric cystoscope for stone removal during open CDC excision. METHODS: All children with CDC presenting with IHSs between January 2015 and December 2017 were included in the present study. IHSs were diagnosed by ultrasound/magnetic resonance cholangiopancreatography (MRCP). In these patients, after cyst excision by open technique, a 9 Fr paediatric cystoscope with 4 Fr working channel was inserted into the common hepatic duct for visualisation and clearance of stones from (intrahepatic bile ducts). Follow-up was done using liver function tests, ultrasound and MRCP (if needed). Patients underwent three monthly liver function test and ultrasound and if needed MRCP. RESULTS: Six cases of CDC presenting with IHS were managed, and one case with post-R-en-Y IHS was treated with this technique. Rigid paediatric cystoscope with working channel and forceps was used. All cases were successfully managed, and one case was found to have intrahepatic duct stenosis was dilated. CONCLUSION: Per-operative rigid endoscopy using paediatric cystoscope is an easily available tool in most of the setups for the management of IHS associated with CDC in children.

Hepatolithiasis: Epidemiology, presentation, classification and management of a complex disease.

The term hepatolithiasis describes the presence of biliary stones within the intrahepatic bile ducts, above the hilar confluence of the hepatic ducts. The disease is more prevalent in Asia, mainly owing to socioeconomic and dietary factors, as well as the prevalence of biliary parasites. In the last century, owing to migration, its global incidence has increased. The main pathophysiological mechanisms involve cholangitis, bile infection and biliary strictures, creating a self-sustaining cycle that perpetuates the disease, frequently characterised by recurrent episodes of bacterial infection referred to as syndrome of "recurrent pyogenic cholangitis". Furthermore, long-standing hepatolithiasis is a known risk factor for development of intrahepatic cholangiocarcinoma. Various classifications have aimed at providing useful insight of clinically relevant aspects and guidance for treatment. The management of symptomatic patients and those with complications can be complex, and relies upon a multidisciplinary team of hepatologists, endoscopists, interventional radiologists and hepatobiliary surgeons, with the main goal being to offer relief from the clinical presentations and prevent the development of more serious complications. This comprehensive review provides insight on various aspects of hepatolithiasis, with a focus on epidemiology, new evidence on pathophysiology, most important clinical aspects, different classification systems and contemporary management.

Evidence-based clinical practice guidelines for cholelithiasis 2021.

The Japanese Society of Gastroenterology first published evidence-based clinical practice guidelines for cholelithiasis in 2010, followed by a revision in 2016. Currently, the revised third edition was published to reflect recent evidence on the diagnosis, treatment, and prognosis of cholelithiasis conforming to the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) system. Following this revision, the present English version of the guidelines was updated and published herein. The clinical questions (CQ) in the previous version were reviewed and rearranged into three newly divided categories: background questions (BQ) dealing with basic background knowledge, CQ, and future research questions (FRQ), which refer to issues that require further accumulation of evidence. Finally, 52 questions (29 BQs, 19 CQs, and 4 FRQs) were adopted to cover the epidemiology, pathogenesis, diagnosis, treatment, complications, and prognosis. Based on a literature search using MEDLINE, Cochrane Library, and Igaku Chuo Zasshi databases for the period between 1983 and August 2019, along with a manual search of new information reported over the past 5 years, the level of evidence was evaluated for each CQ. The strengths of recommendations were determined using the Delphi method by the committee members considering the body of evidence, including benefits and harms, patient preference, and cost-benefit balance. A comprehensive flowchart was prepared for the diagnosis and treatment of gallbladder stones, common bile duct stones, and intrahepatic stones, respectively. The current revised guidelines are expected to be of great assistance to gastroenterologists and general physicians in making decisions on contemporary clinical management for cholelithiasis patients.

The Role of Cholangioscopy in Biliary Diseases.

Endoscopy plays a central role in diagnostic and therapeutic approaches to biliary disease in both benign and malignant conditions. A cholangioscope is an endoscopic instrument that allows for the direct exploration of the biliary tree. Over the years, technology has improved endoscopic image quality and allowed for the development of an operative procedure that can be performed during cholangioscopy. Different types of instruments are available in this context, and they can be used in different anatomical access points according to the most appropriate clinical indication. The direct visualization of biliary mucosa is essential in the presence of biliary strictures of unknown significance, allowing for the appropriate allocation of patients to surgery or conservative treatments. Cholangioscopy has demonstrated excellent performance in discriminating malignant conditions (such as colangiocarcinoma) from benign inflammatory strictures, and more recent advances (e.g., artificial intelligence and confocal laser endomicroscopy) could further increase its diagnostic accuracy. Cholangioscopy also plays a primary role in the treatment of benign conditions such as difficult bile stones (DBSs). In this case, it may not be possible to achieve complete biliary drainage using standard ERCP. Therapeutic cholangioscopy-guided lithotripsy allows for stone fragmentation and complete biliary drainage. Indeed, other complex clinical situations, such as patients with intra-hepatic lithiasis and patients with an altered anatomy, could benefit from the therapeutic role of cholangioscopy. The aim of the present review is to explore the most recent diagnostic and therapeutic advances in the roles of cholangioscopy in the management of biliary diseases.

Diagnosis and management of hepatolithiasis in an adult patient: A case report.

Hepatolithiasis or intrahepatic calculi are common in South East Asia but are rare in Western nations. The primary symptom of the condition is recurrent pain in the upper abdomen. Stones in the cystic duct or common bile duct are also common findings. Recurrent pyogenic cholangitis is the most frequent complication. Radiological studies and percutaneous procedures are vital for diagnosing and managing this condition. The primary goal in treating the condition is to decrease the chance of developing cholangitis and to stop the progression of the disease, which may lead to biliary cirrhosis.

The Difficult Path to Correct Diagnosis of Hepatolithiasis: A Case Report.

Hepatolithiasis is a benign disease, where stones are localized proximal to the confluence of hepatic ducts. The clinical picture may differ depending on whether the stones cause complete, partial, or intermittent biliary obstruction. The course can vary from asymptomatic to fatal, thus, early diagnosis and treatment is critical for a good prognosis. The gold standard in imaging is magnetic resonance cholangiopancreatography (MRCP). However, correct diagnosis can be challenging due to atypical clinical picture and laboratory findings. We present a case where hepatolithiasis was misdiagnosed initially due to incomplete reporting and documentation of MRCP. Choledocholithiasis was diagnosed based on initial MRCP, and endoscopic stone extraction was indicated. However, an unusual post-interventional course and signs of obstructive cholangitis led to an endoscopic re-intervention, which confirmed absence of pathology in extrahepatic biliary ducts. The cholangitis recurrence required intensive antibiotic treatment, and CT examination revealed intrahepatic S3 bile duct dilatation. Thus, a re-evaluation of initial MRCP and repeated MRCP confirmed hepatolithiasis. Further, laparoscopic bisegmentectomy was chosen as the definitive treatment due to the location of the lesion. The patient recovered and remained symptom free upon a 12 month follow up.

Surgical resection for non-Asian intrahepatic lithiasis: The Brazilian experience.

BACKGROUND: Intrahepatic lithiasis (IHL) is a rare disease in the western world. Complications associated with IHL include acute cholangitis, liver atrophy, secondary biliary cirrhosis, and risk for intrahepatic cholangiocarcinoma. Liver resection is considered the treatment of choice for IHL. The objective of this study was to analyze patients who underwent liver resection for non-Asian hepatolithiasis. METHODS: 127 patients with symptomatic non-Asian hepatolithiasis underwent resection in six institutions. Demographic data, clinical presentation, diagnosis, classification according to stone location, presence of atrophy, bile duct stricture, biliary cirrhosis, incidence of cholangiocarcinoma, treatment and postoperative course were evaluated. RESULTS: 52 patients (40.9%) were male and the mean age was 46.1 years. Sixty-six patients (51.9%) presented with history of cholangitis. Stones were located in the left lobe in 63 (49.6%), and right lobe in 28 patients (22.0%). Atrophy was observed in 31 patients (24.4%) and biliary stenosis in 18 patients (14.1%). The most common procedure performed was left lateral sectionectomy in 63 (49.6%) patients, followed by left hepatectomy in 36 (28.3%), right hepatectomy in 19 (15.0%), and associated hepaticojejunostomy in 28 (22.0%). Forty-two patients (33.0%) presented postoperative complications and the most common were biliary fistula (13.3%) and surgical site infection (7.0%). Postoperative mortality was 0.7%. Intrahepatic cholangiocarcinoma was observed in 2 patients (1.5%). Recurrence was identified in 10 patients (7.8%), mostly with bilateral stones and/or hepaticojejunostomy. CONCLUSION: Liver resection is the standard treatment for symptomatic unilateral or complicated IHL with good operative results. Risk of cholangiocarcinoma was low in non-Asian patients.

hsa-miR-93 regulates MUCIN family gene expression via WNT/ß-catenin pathway in intrahepatic stone disease.

BACKGROUND: Mucin plays an essential role in the intrahepatic stone formation, but the mechanism of mucin regulation is unclear. OBJECTIVE: To investigate the potential implication of miR-93 and WNT pathway in the regulation of intrahepatic bile duct mucin expression. METHODS: Thirty patients with or without intrahepatic bile duct stones are involved; Reverse-transcription polymerase chain reaction was performed to evaluate the expression of MUC3, MUC4, MUC5B, MUC5AC mRNA and miR-93 levels. miR-NC or miR-93 mimics was transfected into intrahepatic biliary epithelial cells. Then mucins and Wnt pathway proteins were detected by the immunoblotting, and the target gene TCF7 were validated using the dual luciferase assay. ß-catenin, wnt4, and mucins were an immunohistochemical stain of the intrahepatic biliary epithelial tissues. RESULTS: The expression levels of MUC3, MUC4, MUC5B, and MUC5AC in patients with intrahepatic bile duct stones are higher than control, as well as Wnt pathway proteins (especially ß-catenin and wnt4). Mucins levels increased in wnt4, wnt5a or SB216763-treated HIBECs, and reduced by miR-93 mimics transfection. miR-93 directly targeted TCF7 and repressed Wnt pathway protein expression, which reversed the upregulation of mucin levels induced by wnt4 or wnt5a, but not SB216763. CONCLUSION: These results suggest a new potential mechanism in intrahepatic stones, regulating by miR-93/TCF7, non-canonical Wnt pathway, and mucins.