Introduction: Immune checkpoint inhibitors (ICIs) are widely used to treat various cancers but can induce immune-related adverse events (irAEs) in patients. Herein, we report the case of a 68-year-old patient with hepatocellular carcinoma who developed an overlapping syndrome of irAEs after receiving ICIs.
Case presentation: The patient received transcatheter arterial chemoembolization for hepatocellular carcinoma, followed by immunotherapy of 200 mg of camrelizumab every 3 weeks and 12 mg of lenvatinib mesylate daily. After receiving the second dose of camrelizumab treatment, the patient developed gradually worsening left eyelid ptosis and general weakness. One week later, the patient developed bilateral eyelid ptosis and suffocated while drinking water. Unfortunately, the patient was eventually diagnosed as ICI-related myocarditis, myositis, transaminitis, and myasthenia gravis based on a series of physical examinations. Subsequently, the patient was actively provided life support and symptomatic treatment. His symptoms greatly improved after treatment with methylprednisolone sodium succinate 40 mg once daily intravenously for 7 days, with gradually decreasing doses.
Conclusions: IrAEs are common after ICI treatment. After ICI treatment initiation, close monitoring, early detection, and treatment of irAEs is required to improve clinical outcomes.
{"title":"Overlapping Syndrome of Myocarditis, Myositis, Transaminitis, and Myasthenia Gravis in a Patient with Hepatocellular Carcinoma after Treatment with Immune Checkpoint Inhibitors: A Case Report and Literature Review.","authors":"Baheti Kalifu, Yuan Meng, Kedeerya Aishanjiang, Chao Ma, Guang-Lei Tian, Jin-Guo Wang, Zhi-Gang Ma, Xiong Chen","doi":"10.1159/000547066","DOIUrl":"10.1159/000547066","url":null,"abstract":"<p><strong>Introduction: </strong>Immune checkpoint inhibitors (ICIs) are widely used to treat various cancers but can induce immune-related adverse events (irAEs) in patients. Herein, we report the case of a 68-year-old patient with hepatocellular carcinoma who developed an overlapping syndrome of irAEs after receiving ICIs.</p><p><strong>Case presentation: </strong>The patient received transcatheter arterial chemoembolization for hepatocellular carcinoma, followed by immunotherapy of 200 mg of camrelizumab every 3 weeks and 12 mg of lenvatinib mesylate daily. After receiving the second dose of camrelizumab treatment, the patient developed gradually worsening left eyelid ptosis and general weakness. One week later, the patient developed bilateral eyelid ptosis and suffocated while drinking water. Unfortunately, the patient was eventually diagnosed as ICI-related myocarditis, myositis, transaminitis, and myasthenia gravis based on a series of physical examinations. Subsequently, the patient was actively provided life support and symptomatic treatment. His symptoms greatly improved after treatment with methylprednisolone sodium succinate 40 mg once daily intravenously for 7 days, with gradually decreasing doses.</p><p><strong>Conclusions: </strong>IrAEs are common after ICI treatment. After ICI treatment initiation, close monitoring, early detection, and treatment of irAEs is required to improve clinical outcomes.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"611-620"},"PeriodicalIF":0.6,"publicationDate":"2025-08-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12488183/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145211943","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Introduction: Here, we report a case of gastric cancer associated with autosomal dominant polycystic kidney disease (ADPKD).
Case presentation: A 73-year-old woman with chronic renal failure due to polycystic kidneys, who was undergoing hemodialysis treatment three times a week for 1 month, was admitted to our hospital because of anemia. She reported that her mother had suffered from polycystic kidney disease. Gastrointestinal fiberscopy revealed a type 1 cancerous lesion in the lesser curvature of the antrum. Histopathological analysis of gastric tumor biopsy specimens revealed differentiated tubular adenocarcinoma. Abdominal computed tomography revealed multiple cysts in the liver, cystic lesions on both kidneys, and thickening of the gastric wall. A laparoscopic distal gastrectomy with lymph node dissection was performed.
Conclusion: Reports of the association between gastric cancer and ADPKD are exceptionally uncommon. Especially, no cases of laparoscopic distal gastrectomy for gastric cancer with ADPKD have been previously reported. To our knowledge, this is the first report of laparoscopic surgery for gastric cancer with ADPKD and multiple liver cysts.
{"title":"A Case Report of Laparoscopic Distal Gastrectomy for Gastric Cancer with Autosomal Dominant Polycystic Kidney Disease and Multiple Liver Cysts.","authors":"Yukihiro Kato, Hanae Matsuda, Takuya Kikukawa, Atsushi Isihara, Yasunori Matsuda, Seikan Miyashita, Masahiro Nishikawa, Takahiro Uenishi","doi":"10.1159/000547068","DOIUrl":"10.1159/000547068","url":null,"abstract":"<p><strong>Introduction: </strong>Here, we report a case of gastric cancer associated with autosomal dominant polycystic kidney disease (ADPKD).</p><p><strong>Case presentation: </strong>A 73-year-old woman with chronic renal failure due to polycystic kidneys, who was undergoing hemodialysis treatment three times a week for 1 month, was admitted to our hospital because of anemia. She reported that her mother had suffered from polycystic kidney disease. Gastrointestinal fiberscopy revealed a type 1 cancerous lesion in the lesser curvature of the antrum. Histopathological analysis of gastric tumor biopsy specimens revealed differentiated tubular adenocarcinoma. Abdominal computed tomography revealed multiple cysts in the liver, cystic lesions on both kidneys, and thickening of the gastric wall. A laparoscopic distal gastrectomy with lymph node dissection was performed.</p><p><strong>Conclusion: </strong>Reports of the association between gastric cancer and ADPKD are exceptionally uncommon. Especially, no cases of laparoscopic distal gastrectomy for gastric cancer with ADPKD have been previously reported. To our knowledge, this is the first report of laparoscopic surgery for gastric cancer with ADPKD and multiple liver cysts.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"605-610"},"PeriodicalIF":0.6,"publicationDate":"2025-08-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503541/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249795","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-08-26eCollection Date: 2025-01-01DOI: 10.1159/000547521
Nagarajan Venkatchocku Ashok Annamalai, Garrett Kang, Marianne Anastasia De Roza
Introduction: Acute-on-chronic liver failure (ACLF) associated with multi-organ failure has a high mortality burden. The mechanisms are thought to be driven by hyperactive inflammatory and immune responses triggered by microbial and nonmicrobial factors. Therapeutic plasma exchange (TPE) is a potential alternative to liver transplant that improves survival.
Case presentation: A 56-year-old man with Child-Pugh C11 alcoholic liver cirrhosis was admitted to the intensive care unit for grade 3 ACLF, precipitated by severe alcoholic hepatitis (albumin 29, bilirubin 414, ALP 133, ALT 40, AST 128, Na 123, creatinine 487, INR 1.82) with MELD score of 39. He required inotropic support and renal replacement therapy. The patient was started on antibiotics and steroids for Maddrey Discriminant Function (MDF) score of 67.9. However, there was no improvement with Lille score 0.958 (<0.45) on day 7; hence, steroids were discontinued. The patient was not a liver transplant candidate due to alcoholism and poor social support. He was commenced on a trial of TPE and completed 3 cycles with standard volume plasma replacement resulting in clinical and biochemical improvement. He was discharged from hospital with 90-day survival at outpatient review.
Conclusion: This highlights that TPE is a potential treatment option in alcoholic ACLF patients who would otherwise be at high risk of mortality at 1 month. TPE reduces systemic inflammatory reactions and modulates the adaptive immune response. This is especially important given organ scarcity and limited transplant options for those without social support.
急性慢性肝衰竭(ACLF)与多器官衰竭相关,具有很高的死亡率负担。这种机制被认为是由微生物和非微生物因素引发的过度活跃的炎症和免疫反应驱动的。治疗性血浆交换(TPE)是肝移植的潜在替代方案,可提高生存率。病例介绍:一名56岁男性Child-Pugh C11酒精性肝硬化患者因重度酒精性肝炎(白蛋白29,胆红素414,ALP 133, ALT 40, AST 128, Na 123,肌酐487,INR 1.82)导致的3级ACLF入住重症监护病房,MELD评分为39。他需要肌力支持和肾脏替代治疗。Maddrey判别函数(MDF)评分为67.9,患者开始使用抗生素和类固醇。然而,里尔评分为0.958,没有任何改善(结论:这突出表明TPE是酒精性ACLF患者的潜在治疗选择,否则1个月时死亡风险很高。TPE可减少全身炎症反应并调节适应性免疫反应。鉴于器官稀缺和缺乏社会支持的人的移植选择有限,这一点尤其重要。
{"title":"A Case Report on Plasma Exchange for Acute-on-Chronic Liver Failure from Severe Alcoholic Hepatitis with Failed Steroid Therapy.","authors":"Nagarajan Venkatchocku Ashok Annamalai, Garrett Kang, Marianne Anastasia De Roza","doi":"10.1159/000547521","DOIUrl":"10.1159/000547521","url":null,"abstract":"<p><strong>Introduction: </strong>Acute-on-chronic liver failure (ACLF) associated with multi-organ failure has a high mortality burden. The mechanisms are thought to be driven by hyperactive inflammatory and immune responses triggered by microbial and nonmicrobial factors. Therapeutic plasma exchange (TPE) is a potential alternative to liver transplant that improves survival.</p><p><strong>Case presentation: </strong>A 56-year-old man with Child-Pugh C11 alcoholic liver cirrhosis was admitted to the intensive care unit for grade 3 ACLF, precipitated by severe alcoholic hepatitis (albumin 29, bilirubin 414, ALP 133, ALT 40, AST 128, Na 123, creatinine 487, INR 1.82) with MELD score of 39. He required inotropic support and renal replacement therapy. The patient was started on antibiotics and steroids for Maddrey Discriminant Function (MDF) score of 67.9. However, there was no improvement with Lille score 0.958 (<0.45) on day 7; hence, steroids were discontinued. The patient was not a liver transplant candidate due to alcoholism and poor social support. He was commenced on a trial of TPE and completed 3 cycles with standard volume plasma replacement resulting in clinical and biochemical improvement. He was discharged from hospital with 90-day survival at outpatient review.</p><p><strong>Conclusion: </strong>This highlights that TPE is a potential treatment option in alcoholic ACLF patients who would otherwise be at high risk of mortality at 1 month. TPE reduces systemic inflammatory reactions and modulates the adaptive immune response. This is especially important given organ scarcity and limited transplant options for those without social support.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"597-604"},"PeriodicalIF":0.6,"publicationDate":"2025-08-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503534/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249765","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-08-20eCollection Date: 2025-01-01DOI: 10.1159/000546558
Landen Shane Burstiner, Yasasvhinie Santharam, Christopher Mikulas, Avni Agrawal, Reshmi Mathew, Peter Senada, Maged Peter Ghali
Introduction: Kratom is an herbal preparation - generally an extract, capsule, chew, or smoking substrate - made from the leaves of a tropical tree (Mitragyna speciosa) that is indigenous to Southeast Asia. Its physiological effects are complex and dose dependent: low doses mimic stimulants, while higher doses have sedative and analgesic effects. Although injury and failure of various organs have been reported with kratom use across multiple case study analyses, gastrointestinal involvement has been rarely documented.
Case presentation: We present a 26-year-old female with no past medical history, no risk factors for intestinal ischemia, and an addiction to the commercial substance, kratom, who presented for 1 day of bright-red bloody diarrhea and bilious vomiting, and several weeks of intermittent sharp abdominal pain. Colonoscopy showed decreased mucosa vascular pattern in watershed areas of the colon and a splenic flexure that was so ulcerated it could not be safely traversed.
Conclusion: Given the lack of concomitant risk factors and comorbidities, as well as the timing of her symptoms beginning well before she started any medications, our most likely patient developed ischemic colitis due to her kratom use. This is further supported by her repeat colonoscopy 2 months after quitting kratom, which showed complete resolution. We would advise vigilance for possible bowel ischemia in those using kratom, a substance on which there is currently limited knowledge.
{"title":"A Novel Case of Ischemic Colitis in the Setting of Kratom Use: A Case Report.","authors":"Landen Shane Burstiner, Yasasvhinie Santharam, Christopher Mikulas, Avni Agrawal, Reshmi Mathew, Peter Senada, Maged Peter Ghali","doi":"10.1159/000546558","DOIUrl":"10.1159/000546558","url":null,"abstract":"<p><strong>Introduction: </strong>Kratom is an herbal preparation - generally an extract, capsule, chew, or smoking substrate - made from the leaves of a tropical tree (<i>Mitragyna speciosa</i>) that is indigenous to Southeast Asia. Its physiological effects are complex and dose dependent: low doses mimic stimulants, while higher doses have sedative and analgesic effects. Although injury and failure of various organs have been reported with kratom use across multiple case study analyses, gastrointestinal involvement has been rarely documented.</p><p><strong>Case presentation: </strong>We present a 26-year-old female with no past medical history, no risk factors for intestinal ischemia, and an addiction to the commercial substance, kratom, who presented for 1 day of bright-red bloody diarrhea and bilious vomiting, and several weeks of intermittent sharp abdominal pain. Colonoscopy showed decreased mucosa vascular pattern in watershed areas of the colon and a splenic flexure that was so ulcerated it could not be safely traversed.</p><p><strong>Conclusion: </strong>Given the lack of concomitant risk factors and comorbidities, as well as the timing of her symptoms beginning well before she started any medications, our most likely patient developed ischemic colitis due to her kratom use. This is further supported by her repeat colonoscopy 2 months after quitting kratom, which showed complete resolution. We would advise vigilance for possible bowel ischemia in those using kratom, a substance on which there is currently limited knowledge.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"590-595"},"PeriodicalIF":0.6,"publicationDate":"2025-08-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503479/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249751","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Introduction: Reactive intravascular plasmablastic/immunoblastic proliferation (RIVPIP) is rarely reported. RIVPIP may histologically mimic aggressive intravascular lymphoma, especially in the context of an old patient presenting with concurrent indolent lymphoma and tuberculosis, and easily lead to erroneous diagnosis.
Case presentation: A 79-year-old female presenting with upper abdominal discomfort, weight loss, hypoproteinemia, and polyserosal effusions underwent a gastric endoscopy examination. Biopsy revealed that the lamina propria was infiltrated by a large number of uniformly small-sized lymphocytes (CD20+/PAX5+) with lymphoepithelial lesions and epithelioid granuloma. Notably, vascular lumens were filled and distended by abundant large-sized plasmablasts or immunoblasts (CD79α+/MUM1+/CD20-/CD138-/PAX5-/CD30-/Bcl-2-/Bcl-6-/C-myc-). These intravascular large lymphoid cells demonstrated high proliferative activity (Ki67 >80%) without immunoglobulin light chain restriction or EBV association (EBER-ISH negative). Furthermore, PCR revealed monoclonal rearrangements in IgH, IgK, and IgL genes. The patient had a history of endoscopic gastric biopsy 2 months ago. Retrospective analysis of a prior biopsy identified conspicuous caseating necrosis, epithelioid granuloma, multinucleated giant cells with suspicious positivity for acid-fast staining, highly suggestive of tuberculosis. Based on two biopsy specimens and systemic symptoms, the case was finally diagnosed as gastric MALT lymphoma and tuberculosis with extensive RIVPIP.
Conclusion: We present an unusual case of RIVPIP with severe systemic symptoms due to concurrent gastric MALT lymphoma and tuberculosis, emphasizing a potential diagnostic pitfall for clinical pathologists in distinguishing RIVPIP from the aggressive intravascular lymphoma. A comprehensive evaluation of clinical manifestations, pathological morphology, immunophenotype and gene analysis is required to make the precise diagnosis.
{"title":"Reactive Intravascular Plasmablastic/Immunoblastic Proliferation in a Patient with Concurrent Gastric Mucosa-Associated Lymphoid Tissue Lymphoma and Tuberculosis Potentially Mimicking Aggressive Intravascular Lymphoma: A Rare Case Report.","authors":"Ling Ling Zhong, Zhi Ping Tang, Hai Peng Zhang, Shuang Zhong, Ying Wen Huang, Gao Xiang Huang","doi":"10.1159/000547190","DOIUrl":"10.1159/000547190","url":null,"abstract":"<p><strong>Introduction: </strong>Reactive intravascular plasmablastic/immunoblastic proliferation (RIVPIP) is rarely reported. RIVPIP may histologically mimic aggressive intravascular lymphoma, especially in the context of an old patient presenting with concurrent indolent lymphoma and tuberculosis, and easily lead to erroneous diagnosis.</p><p><strong>Case presentation: </strong>A 79-year-old female presenting with upper abdominal discomfort, weight loss, hypoproteinemia, and polyserosal effusions underwent a gastric endoscopy examination. Biopsy revealed that the lamina propria was infiltrated by a large number of uniformly small-sized lymphocytes (CD20+/PAX5+) with lymphoepithelial lesions and epithelioid granuloma. Notably, vascular lumens were filled and distended by abundant large-sized plasmablasts or immunoblasts (CD79α+/MUM1+/CD20-/CD138-/PAX5-/CD30-/Bcl-2-/Bcl-6-/C-myc-). These intravascular large lymphoid cells demonstrated high proliferative activity (Ki67 >80%) without immunoglobulin light chain restriction or EBV association (EBER-ISH negative). Furthermore, PCR revealed monoclonal rearrangements in IgH, IgK, and IgL genes. The patient had a history of endoscopic gastric biopsy 2 months ago. Retrospective analysis of a prior biopsy identified conspicuous caseating necrosis, epithelioid granuloma, multinucleated giant cells with suspicious positivity for acid-fast staining, highly suggestive of tuberculosis. Based on two biopsy specimens and systemic symptoms, the case was finally diagnosed as gastric MALT lymphoma and tuberculosis with extensive RIVPIP.</p><p><strong>Conclusion: </strong>We present an unusual case of RIVPIP with severe systemic symptoms due to concurrent gastric MALT lymphoma and tuberculosis, emphasizing a potential diagnostic pitfall for clinical pathologists in distinguishing RIVPIP from the aggressive intravascular lymphoma. A comprehensive evaluation of clinical manifestations, pathological morphology, immunophenotype and gene analysis is required to make the precise diagnosis.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"581-589"},"PeriodicalIF":0.6,"publicationDate":"2025-08-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503462/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249961","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-08-16eCollection Date: 2025-01-01DOI: 10.1159/000546532
Isa N van der Laan, Marjolein Y V Homs, Eva V E Madsen, Willem J Lammers
Introduction: Endoscopic ultrasound-guided gastroenterostomy is an emerging treatment for malignant gastric outlet obstruction, with high clinical and technical success rates and reported complication risks around 10%. However, the long-term risks associated with the presence of a metal stent remain unclear.
Case presentation: We present the case of a 35-year-old male with duodenal carcinoma and peritoneal metastases, treated with endoscopic ultrasound-guided gastroenterostomy because of gastric outlet obstruction, who developed a delayed jejunal perforation 10 months after the procedure. The perforation, likely related to the lumen-apposing metal stent, was resected. Thereafter, the patient recovered and resumed immunotherapy with no measurable disease on follow-up computed tomography.
Conclusion: This report describes a case with a delayed jejunal perforation 10 months after endoscopic ultrasound-guided gastroenterostomy in a patient with metastatic duodenal carcinoma with complete treatment response to immunotherapy. We advise stent removal once the duodenal obstruction has resolved, given the potential risk of complications associated with prolonged stents in place.
{"title":"Delayed Jejunal Perforation after Endoscopic Ultrasound-Guided Gastroenterostomy: A Case Report and Literature Review.","authors":"Isa N van der Laan, Marjolein Y V Homs, Eva V E Madsen, Willem J Lammers","doi":"10.1159/000546532","DOIUrl":"10.1159/000546532","url":null,"abstract":"<p><strong>Introduction: </strong>Endoscopic ultrasound-guided gastroenterostomy is an emerging treatment for malignant gastric outlet obstruction, with high clinical and technical success rates and reported complication risks around 10%. However, the long-term risks associated with the presence of a metal stent remain unclear.</p><p><strong>Case presentation: </strong>We present the case of a 35-year-old male with duodenal carcinoma and peritoneal metastases, treated with endoscopic ultrasound-guided gastroenterostomy because of gastric outlet obstruction, who developed a delayed jejunal perforation 10 months after the procedure. The perforation, likely related to the lumen-apposing metal stent, was resected. Thereafter, the patient recovered and resumed immunotherapy with no measurable disease on follow-up computed tomography.</p><p><strong>Conclusion: </strong>This report describes a case with a delayed jejunal perforation 10 months after endoscopic ultrasound-guided gastroenterostomy in a patient with metastatic duodenal carcinoma with complete treatment response to immunotherapy. We advise stent removal once the duodenal obstruction has resolved, given the potential risk of complications associated with prolonged stents in place.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"574-580"},"PeriodicalIF":0.6,"publicationDate":"2025-08-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503476/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249874","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-08-14eCollection Date: 2025-01-01DOI: 10.1159/000545824
Alice Leah Kerkham, Martin David Weltman, Zoe Jennings, Han Liem
Introduction: Disseminated histoplasmosis is a rare cause of granulomatous, cholestatic liver disease. It can also manifest with gastrointestinal (GI) tract involvement, usually with ulceration or mucosal thickening, but is rarely reported as a cause of mass lesions in the GI tract. It is also very rare for disseminated histoplasmosis to present in an immunocompetent host.
Case presentation: We present a case of cholestatic liver disease and a small bowel mass in a 50-year-old male that has not previously been described in an immunocompetent host.
Conclusion: Timely diagnosis requires a high index of suspicion, particularly if there has been exposure to high-risk environments such as contaminated soils, as delayed diagnosis is associated with significant mortality.
{"title":"Disseminated Histoplasmosis as a Cause of a Small Bowel Mass and Cholestatic Liver Injury in an Immunocompetent Host: A Case Report.","authors":"Alice Leah Kerkham, Martin David Weltman, Zoe Jennings, Han Liem","doi":"10.1159/000545824","DOIUrl":"10.1159/000545824","url":null,"abstract":"<p><strong>Introduction: </strong>Disseminated histoplasmosis is a rare cause of granulomatous, cholestatic liver disease. It can also manifest with gastrointestinal (GI) tract involvement, usually with ulceration or mucosal thickening, but is rarely reported as a cause of mass lesions in the GI tract. It is also very rare for disseminated histoplasmosis to present in an immunocompetent host.</p><p><strong>Case presentation: </strong>We present a case of cholestatic liver disease and a small bowel mass in a 50-year-old male that has not previously been described in an immunocompetent host.</p><p><strong>Conclusion: </strong>Timely diagnosis requires a high index of suspicion, particularly if there has been exposure to high-risk environments such as contaminated soils, as delayed diagnosis is associated with significant mortality.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"568-573"},"PeriodicalIF":0.6,"publicationDate":"2025-08-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503467/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249883","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Introduction: Enterocytozoon bieneusi (E.b) can cause opportunistic infections in immunocompromised patients, especially HIV/AIDS recipients, but currently, there are still few case reports of E.b infections in solid organ transplant recipients. As E.b cannot be diagnosed by routine examination and culture, it is often overlooked as a possible cause of chronic diarrhea in transplant recipients. With no known established guidelines for E.b, the effective and available treatment options are limited.
Case presentation: In this article, we reported a case of persistent chronic diarrhea and renal failure due to E.b infection after liver transplantation, which was definitively diagnosed using metagenomic next-generation sequencing (mNGS). Short course of albendazole was applied to the patient, resulting in rapid resolution of clinical symptoms and recovery of renal function.
Conclusion: The case demonstrates the advantages of mNGS for the diagnosis of rare pathogenic bacterial infections, and together with the previous case reports, further suggests albendazole may serve as an effective therapy for the treatment of E.b for some patient. This article reviews relevant literature and provides an updated and more comprehensive reference for the selection of E.b treatment drugs. To be note, the overview showed there may be individualized differences in the efficacy of albendazole, which needs more study.
{"title":"Infection of <i>Enterocytozoon bieneusi</i> in a Patient with Long-Term Immunosuppression after Liver Transplantation: A Case Report and Literature Review.","authors":"Xiaohua Hong, Lei Zeng, Qiong Li, Tiankai Yang, Jing Zhou, Huayou Li, Yunchao Mo, Shaoyi Zhuo, Jiudong Ma, Jingyue Cai, Caipeng Xie","doi":"10.1159/000546660","DOIUrl":"10.1159/000546660","url":null,"abstract":"<p><strong>Introduction: </strong><i>Enterocytozoon bieneusi</i> (<i>E.b</i>) can cause opportunistic infections in immunocompromised patients, especially HIV/AIDS recipients, but currently, there are still few case reports of <i>E.b</i> infections in solid organ transplant recipients. As <i>E.b</i> cannot be diagnosed by routine examination and culture, it is often overlooked as a possible cause of chronic diarrhea in transplant recipients. With no known established guidelines for <i>E.b</i>, the effective and available treatment options are limited.</p><p><strong>Case presentation: </strong>In this article, we reported a case of persistent chronic diarrhea and renal failure due to <i>E.b</i> infection after liver transplantation, which was definitively diagnosed using metagenomic next-generation sequencing (mNGS). Short course of albendazole was applied to the patient, resulting in rapid resolution of clinical symptoms and recovery of renal function.</p><p><strong>Conclusion: </strong>The case demonstrates the advantages of mNGS for the diagnosis of rare pathogenic bacterial infections, and together with the previous case reports, further suggests albendazole may serve as an effective therapy for the treatment of <i>E.b</i> for some patient. This article reviews relevant literature and provides an updated and more comprehensive reference for the selection of <i>E.b</i> treatment drugs. To be note, the overview showed there may be individualized differences in the efficacy of albendazole, which needs more study.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"556-567"},"PeriodicalIF":0.6,"publicationDate":"2025-08-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12334147/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144815815","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-08-07eCollection Date: 2025-01-01DOI: 10.1159/000546485
Ankita Nekkanti, Harishankar Gopakumar, Muhammad Asghar, Manasa Kandula, Srinivas Puli
Introduction: Hepatic cysts, commonly discovered incidentally, can become symptomatic due to complications like infection, rupture, or mass effect. Traditional management options include percutaneous drainage, open surgical or laparoscopic deroofing, and liver resection. We present a case series of 3 patients with complex fluid collections in the liver managed by endoscopic ultrasound (EUS)-guided drainage using lumen-apposing metal stent (LAMS) placement. While reports exist from other countries, our study contributes a series of cases from the USA.
Case presentation: Three patients, average age 75 years (two men, one woman), underwent EUS-guided drainage using LAMS for an infected liver cyst, infected liver hematoma within a hepatocellular carcinoma lesion, and a symptomatic simple liver cyst, respectively. All the hepatic fluid collections were in the left hepatic lobe and had an average diameter of 11 centimeters (cm). Only the female patient with a symptomatic cyst had prior percutaneous drainage. Successful LAMS placement was achieved in all cases. Clinical success, defined as cyst resolution or significant size reduction, was observed in all patients. The superinfected liver cyst showed complete resolution, yet the stent remained in place as the patient transitioned to hospice and subsequently passed away. In the case of the superinfected liver hematoma, the stent was removed after 2 months. No recurrence was observed in follow-up imaging 8 months later, and the patient passed away. The symptomatic simple liver cyst patient had the stent removed 15 months later, with no recurrence in imaging 11 months of post-stent removal.
Conclusion: EUS-guided LAMS placement emerges as a less invasive and viable option for treating symptomatic and/or infected hepatic fluid collections compared to surgery or percutaneous drainage. While it may be a preferable choice in institutions with the requisite expertise, further studies are essential to establish its definitive role as a first-line intervention.
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