Primary mucoepidermoid carcinoma of the intrahepatic bile duct: A case report.
Ontology highlight
ABSTRACT:
SUBMITTER: Kato K
PROVIDER: S-EPMC8811179 | biostudies-literature | 2022 Feb
REPOSITORIES: biostudies-literature
Publications
Primary mucoepidermoid carcinoma of the intrahepatic bile duct: A case report.
Kato Kenichi K Hara Yasuyuki Y Nishida Akiko A Suzuki Shuhei S Sato Hidenori H Chin Masahiro M Hashizume Eiji E
Clinical case reports 20220202 2
Mucoepidermoid carcinoma (MEC) is the most common salivary gland carcinoma; however, hepatobiliary MEC is extremely rare. A 74-year-old patient was diagnosed with hepatobiliary MEC after hepatectomy. We considered its origin could be the peribiliary glands. Its genome profile was similar to salivary MEC rather than standard biliary tract carcinoma. ...[more]
Similar Datasets
Project description:Introduction and importanceIntraductal papillary neoplasm of the bile duct (IPNB) is a tumour with a very low incidence in the Western world, characterised by a high risk of malignant transformation and unknown prognosis. It is a new entity which was adopted by the WHO in 2010 as a precursor lesion of cholangiocarcinoma. Intrahepatic bile duct is the most common site of origin for IPNB.Case presentationHereby, we present a case of an asymptomatic 63- year-old man, referred to our department after routine ultrasonography showing a multifocal cystic lesion on the left hepatic lobe. Further screening modalities (CT, MRI abdo) confirmed a complex cystic liver lesion with atypical features. The patient underwent left hepatectomy. Histopathology showed a cystic type intrahepatic IPNB, which was completely resected (R0). The follow up in 2 yrs post-operation showed no signs of recurrence.Clinical discussionThe diagnosis and management of IPNB remain challenging. A multimodality imaging approach is essential in order to diagnose IPNB, assess tumour location and extent and plan the optimal treatment strategy.ConclusionComplete surgical resection (R0) with close postoperative follow-up offers long-term survival.
| S-EPMC7900680 | biostudies-literature
Project description: Not available
| S-EPMC8460092 | biostudies-literature
Project description:BackgroundRaoultella Planticola is a facultative anaerobic, gram-negative, water- and soil-dwelling rod bacterium rarely reported as a cause of human disease. However, the number of reported R. planticola infections is growing, without a concomitant increase in research on the microbe or its pathogenesis. Previous genomic studies demonstrating genetic similarities between R. planticola and Klebsiella pneumoniae suggest that capsule biosynthesis, mucoid phenotype, biofilm production, and lipopolysaccharide (endotoxin) synthesis may all be potential virulence factors of R. planticola. We present a unique case of R. planticola infection of the biliary tract 5 years after biliary surgery in a patient with no previously documented risk factors. We also use in silico techniques to predict virulence factors of R. planticola.Case presentationThis case report is the first to discuss a R. planticola infection in the biliary tract of late onset post-surgery (5 years) in a Caucasian patient with no previously documented risk factors.ConclusionsAn in-depth search of the current literature did not yield other similar cases of R. planticola infections. Moreover, to the best of our knowledge, our case is the first case of R. planticola isolated from post-endoscopic retrograde cholangiopancreatography (ERCP) as part of biliary sepsis not associated with gastroenteritis. The late onset of the infection in our patient and the results of the in silico analysis suggest that R. planticola may have survived exposure to the host immune system through the creation of an intracellular biofilm or in a non-culturable but viable state (NCBV) for the 5-year period. The in silico analysis also suggests that biofilms, enterobactin, and mucoid phenotype may play a role in the pathogenesis of R. planticola. However, further research is needed to illuminate the significance of pili, capsule biosynthesis, and lipopolysaccharide (LPS) in the virulence of R. planticola. Lastly, as our patient did not have any risk factors previously associated with R. planticola, we suggest that biliary tract stricture, cholecystitis, and prior surgery may be possible novel risk factors.
| S-EPMC8025561 | biostudies-literature
Project description:Few studies have reported the long-term outcomes of surgical resected intraductal papillary neoplasm of the bile duct (IPNB). Here, we describe the long-term observation and treatment of a case of widespread IPNB.A 57-year-old male was referred to our hospital due to jaundice and dilation of the intrahepatic bile duct. Computed tomography showed dilation and irregularities of the right intrahepatic and extrahepatic bile ducts together with a 3cm nodule in the common hepatic duct. Peroral cholangioscopy revealed mucinous discharge from the ampulla of Vater, which resulted in a diagnosis of IPNB. A biopsy of the nodule and the bile duct revealed papillary adenoma in all of them. Right hepatectomy, caudate lobectomy, extrahepatic bile duct resection, and left hepaticojejunostomy were performed. The nodule was histologically diagnosed as papillary carcinoma in situ, and R0 resection was performed. However, mucus production from the papillary adenoma in the B3 and B4 was observed. We carefully managed the patient's biliary tract by inserting a biliary drainage tube into the segment 2, and he has survived for more than 7 years since the initial treatment.Mucus might be produced after the surgical resection of IPNB even if s surgical margin was benign. Five-year survival rate of benign IPNB was reported from 85% to 100%. That might be caused by difference of the postoperative management of the biliary tract.Careful management of the biliary tract should be performed after surgical resection of IPNB.
| S-EPMC5536823 | biostudies-other
Project description:IntroductionEmbryonal Rhabdomyosarcoma (ERMS) is a malignant soft tissue musculoskeletal tumor which constitutes about 0.06% of all malignancies affecting children. Biliary tract ERMS is still rare, though it is considered the most common cause of malignant obstructive jaundice in children.Case presentationA report of a 2-year-old boy, who was presented with recurrent episodes of scleral icterus of three months duration, is added to the related literature. His labs went with obstructive jaundice and the radiological investigations were consistent with a diagnosis of choledochal cyst. The found mass was suspected to be an ERMS of common bile duct and turned out to be so by the histopathology. He was managed totally by laparoscope, both excision and hepaticojejunostomy reconstruction, which is an extremely uncommon entity.ConclusionCommon Bile Duct Rhabdomyosarcoma is rare and diagnosis at this anatomical site is difficult. Our case highlights the feasibility of laparoscopic resection and hepaticojejunostomy reconstruction with very good results at 16-month follow up and parents' gratitude as well.
| S-EPMC7519210 | biostudies-literature
Project description:BackgroundLymphoepithelioma-like intrahepatic cholangiocarcinoma (LEL-ICC) is a rare variant of intrahepatic cholangiocarcinoma. Epstein-Barr virus (EBV) infection was considered to play a pivotal role in the tumorigenesis of LEL-ICC. It is difficult to diagnosis of LEL-ICC due to the lack of specific features regarding the laboratory test results and imaging findings. At present, the diagnosis of LEL-ICC mainly depends on the histopathologic and immunohistochemical examinations. In addition, the prognosis of LEL-ICC was better than classical cholangiocarcinomas. To our knowledge, only few cases of LEL-ICC have been reported in the literature.Case presentationWe presented a case of a 32-year-old Chinese female with LEL-ICC. She had a 6-month history of upper abdominal pain. The magnetic resonance imaging (MRI) showed a 1.1× 1.3 cm lesion in the left lobe of liver, appearing low signal intensity on T1-weighted images and high signal intensity on T2-weighted images. The patient underwent laparoscopic left lateral sectionectomy. The postoperative histopathologic and immunohistochemical examinations results allowed for the definitive diagnosis of LEL-ICC. The patient was free from tumor recurrence after a 28 months follow-up.ConclusionIn this study, we reported a rare case of LEL-ICC associated with both HBV and EBV infection. EBV infection might play a pivotal role in the carcinogenesis of LEL-ICC, and surgical resection is still the most effective treatment at present. Further research on the etiology and treatment strategies of LEL-ICC is required.
| S-EPMC10183565 | biostudies-literature
Project description:IntroductionGiant duodenal diverticulum is a very rare case. There are only few cases reported. We reported a case of giant duodenal diverticulum with biliary obstruction caused by mucinous carcinoma of distal common bile duct (CBD), that mimicking Lemmel syndrome.Case presentationA 68-years-old man admitted to hospital with recurrent epigastric pain, jaundice and fever. Magnetic resonance cholangiopancreatography showed dilated intrahepatic and extrahepatic biliary tree, dilated gallbladder and cystic mass in pancreatic head that pushed the pancreatic duct ventrally. Emergency laparotomy was performed. Distended edematous gallbladder with necrotic spot, dilated of CBD and compressible bulging of the pancreatic head were found. Duodenotomy in 2nd-3rd part was made and found a giant duodenal diverticulum filled with food and mucus. Tight adhesion to the ampula of Vater, common bile duct, and pancreas due to fibrosis, met difficulties in dissection with a lot of bleeding, hence the diverticulum was not removed. Gastrojejunostomy, cholecystectomy and choledocho-duodenostomy were also done. Pathologic examination of CBD mucus was accordance with mucinous carcinoma.DiscussionPeriampullary duodenal diverticulum can cause obstructive jaundice, known as Lemmel syndrome. This case was different as the giant duodenal diverticulum located in the 3rd part filled with food and mucin that compressed both distal CBD and pancreatic duct. The cause of obstructive jaundice could be fibrotic tissue in distal CBD and mucinous carcinoma.ConclusionGiant duodenal diverticulum with bile obstruction is very rare and challenging in diagnosis and treatment. The other cause of obstruction should be considered such as mucinous carcinoma of distal CBD.
| S-EPMC8760494 | biostudies-literature
Project description:Introduction and importance Primary Gallbladder hydatid cysts are a very rare phenomenon caused by Echinococcus granulosus. Hydatid cysts usually present as hepatic or pulmonary lesions, but in our case, it presented in the gallbladder with three symptomatic daughter cysts in the CBD. Echinococcus caused by E.granulosus Is the most common parasite causing the disease, accounting for 95% of the cases. Case presentation 75 - year - old male presented with colic pain, jaundice, itching, vomiting, nausea, insomnia, Positive murphy's sign, and elevated AST and ALT. Total bilirubin was also elevated with no hepatomegaly or splenomegaly. Clinical discussion This disease is endemic in Mediterranean countries due to high contact with the host of the parasite the lumen or on the external surface of the gallbladder. Which can come from the portal system or the spreading of brood capsules through the biliary tract. Conclusion The method of investigation used was USG, CT, ERCP which are affordable and accessible in low-income countries including Syria. we would like to highlight this rare presentation and the possibility of using laparoscopic surgery. Highlights • Primary hydatid cyst is usually found in the liver, lungs and brain in our case it presented in the gallbladder and common bile duct which is very rare.• We can encounter primary hydatid cyst in unexpected organs especially in countries with high prevalence.• Jaundice in our case was the main presenting symptom.• The role of laparoscopic surgery in treatment because of its less invasive nature and the low spillage possibility. This is the second case treated via laparoscopic surgery.
| S-EPMC9283885 | biostudies-literature
Project description:Primary mucoepidermoid carcinoma of the liver (PMCL) is rare in the hepatic system, with no standard treatment and poor prognosis with a median overall survival of only 120 days. PMCL with immunotherapy has not been reported yet. Here, we present a case of PMCL treated by immunotherapy and chemotherapy. A 64-year-old male with PMCL underwent partial right hepatectomy and liver lesion resection on 19 June 2020. Two months later, the chest computed tomography indicated the presence of multiple nodules in both lungs with higher tumor markers. Considering the presence of a tumor metastasis, the patient received four courses of immunotherapy plus mGEMOX chemotherapy from 8 September 2020. The patient tolerated the combined therapy well, with red moles on the face and chest which were considered as grade 1 reactive cutaneous capillary endothelial proliferation. He also had grade 2 thrombocytopenia and leucopenia after the first course of chemotherapy, but no neutropenia, fatigue, vomiting or diarrhea. However, his disease progressed. The patient refused further treatment and died on 20 April 2021. The overall survival time after diagnosis was 301 days. We describe here the first case report on immunotherapy treatment for PMCL. That suggested immunotherapy combined with chemotherapy may be an option after a hepatic lobectomy for PMCL.
| S-EPMC9997629 | biostudies-literature
Project description:We report a case of intrahepatic bile duct adenoma (BDA) detected during laparoscopic distal gastrectomy for gastric cancer. A 70-year-old man was referred to our hospital for the treatment of gastric cancer. Esophagogastroduodenoscopy revealed an irregular, nodular, and elevated lesion on the greater curvature side of the middle third of the stomach. Abdominal contrast-enhanced computed tomography showed wall thickening with homogeneous enhancement in the middle part of the stomach, and no lesions in the liver. The patient underwent laparoscopic distal gastrectomy with regional lymphadenectomy, and during the operation a small whitish nodule was observed on the lateral segment of the liver surface. The lesion was excised by partial resection of the liver for the purpose of both histological diagnosis and treatment. Pathological examination of the liver lesion revealed no structural or cellular atypia, no stromal invasion, and immunohistochemical positivity for CK7 and CK19, but negativity for p53. The final diagnosis was well-differentiated adenocarcinoma invading the gastric serosal layer without lymph node metastasis, and intrahepatic BDA measuring 0.4?×?0.3 cm. Following surgery, the patient remained symptom-free without evidence of recurrence for 5 months. To the best of our knowledge, this is the first case of BDA with gastric cancer. Because it is difficult to distinguish BDA from other liver tumors including metastatic cancer due to its characteristically small size and lack of specific morphological features on standard imaging, surgical resection should be considered as the most suitable approach for both accurate diagnosis and treatment.
| S-EPMC6498292 | biostudies-literature