Pub Date : 2024-06-27eCollection Date: 2024-01-01DOI: 10.1159/000539646
Harry Wilson, Douglas Macdonald, Kathleen Bryce
Introduction: Patients with advanced hepatocellular carcinoma (HCC) have limited treatment options in the context of decompensated cirrhosis. HCC occurs in patients with hepatitis C virus (HCV) infection and cirrhosis at 1-4% per year. Direct-acting antiviral (DAA) efficacy is decreased in the presence of HCC. We present a case where immunotherapy may have resulted in HCV clearance, when DAA therapy had been ineffective. We hypothesise that immune checkpoint inhibitors targeting the PD-1/PD-L1 pathway can reverse T-cell exhaustion and aid in the clearance of chronic HCV.
Case presentation: This case study describes a male in his 40 s identified by a re-engagement initiative for HCV, who had been unaware of his diagnosis. On further investigation he was found to have compensated for liver cirrhosis and HCC. He was treated with HCV DAA therapy (sofosbuvir/velpatasvir) and then systemic immunotherapy for HCC with atezolizumab and bevacizumab, in an attempt to downstage the disease. Hepatitis C therapy did not achieve sustained virological response, with viral relapse after the end of treatment. This, combined with ongoing alcohol use, resulted in hepatic decompensation and cessation of immunotherapy after the fifth cycle. The HCV RNA subsequently became undetectable without further DAA re-treatment.
Conclusion: To our knowledge, this is the first case of HCV clearance after DAA relapse and the timing of this event after immunotherapy suggests a causal link. We hypothesise that this may be due to the reversal of antiviral T-cell exhaustion. This would therefore support further investigation into other chronic viral infections that create tumour associated with immunosuppressive microenvironments.
{"title":"Clearance of Hepatitis C Virus following Immune Checkpoint Inhibitor Therapy for Hepatocellular Carcinoma: Case Report.","authors":"Harry Wilson, Douglas Macdonald, Kathleen Bryce","doi":"10.1159/000539646","DOIUrl":"10.1159/000539646","url":null,"abstract":"<p><strong>Introduction: </strong>Patients with advanced hepatocellular carcinoma (HCC) have limited treatment options in the context of decompensated cirrhosis. HCC occurs in patients with hepatitis C virus (HCV) infection and cirrhosis at 1-4% per year. Direct-acting antiviral (DAA) efficacy is decreased in the presence of HCC. We present a case where immunotherapy may have resulted in HCV clearance, when DAA therapy had been ineffective. We hypothesise that immune checkpoint inhibitors targeting the PD-1/PD-L1 pathway can reverse T-cell exhaustion and aid in the clearance of chronic HCV.</p><p><strong>Case presentation: </strong>This case study describes a male in his 40 s identified by a re-engagement initiative for HCV, who had been unaware of his diagnosis. On further investigation he was found to have compensated for liver cirrhosis and HCC. He was treated with HCV DAA therapy (sofosbuvir/velpatasvir) and then systemic immunotherapy for HCC with atezolizumab and bevacizumab, in an attempt to downstage the disease. Hepatitis C therapy did not achieve sustained virological response, with viral relapse after the end of treatment. This, combined with ongoing alcohol use, resulted in hepatic decompensation and cessation of immunotherapy after the fifth cycle. The HCV RNA subsequently became undetectable without further DAA re-treatment.</p><p><strong>Conclusion: </strong>To our knowledge, this is the first case of HCV clearance after DAA relapse and the timing of this event after immunotherapy suggests a causal link. We hypothesise that this may be due to the reversal of antiviral T-cell exhaustion. This would therefore support further investigation into other chronic viral infections that create tumour associated with immunosuppressive microenvironments.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"347-351"},"PeriodicalIF":0.5,"publicationDate":"2024-06-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250384/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626097","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 : 2024-06-20eCollection Date: 2024-01-01DOI: 10.1159/000539439
Alex Yartsev, Jessica T Nguyen
Introduction: Although terlipressin is known to cause bradycardia, this adverse effect is usually described in association with hypertension and is considered a benign compensatory response mediated by arterial baroreceptors. Cardiac monitoring for patients receiving terlipressin is not routinely recommended.
Case presentation: A 77-year-old female patient with no history of coronary artery disease and no other coexisting risk factors for cardiac arrhythmias or conduction disturbances was admitted to intensive care unit with severe cholangitis, complicated by variceal bleeding. She developed severe sinus bradycardia following the use of terlipressin, which was associated with significant hypotension that required the infusion of norepinephrine. The bradycardia occurred again when terlipressin therapy was reattempted.
Conclusion: Vasopressin is known to sensitize baroreceptor reflexes by a central mechanism though its actions on V1a receptors in the area postrema, and we speculate that vasopressin analogues such as terlipressin may act in the same manner. That this effect is not widely described in terlipressin safety literature may be due to the overall younger age range of the trial population. This raises the possibility that cardiac monitoring may be warranted for elderly patients receiving terlipressin.
{"title":"Severe Bradycardia Associated with the Use of Terlipressin: A Case Report.","authors":"Alex Yartsev, Jessica T Nguyen","doi":"10.1159/000539439","DOIUrl":"10.1159/000539439","url":null,"abstract":"<p><strong>Introduction: </strong>Although terlipressin is known to cause bradycardia, this adverse effect is usually described in association with hypertension and is considered a benign compensatory response mediated by arterial baroreceptors. Cardiac monitoring for patients receiving terlipressin is not routinely recommended.</p><p><strong>Case presentation: </strong>A 77-year-old female patient with no history of coronary artery disease and no other coexisting risk factors for cardiac arrhythmias or conduction disturbances was admitted to intensive care unit with severe cholangitis, complicated by variceal bleeding. She developed severe sinus bradycardia following the use of terlipressin, which was associated with significant hypotension that required the infusion of norepinephrine. The bradycardia occurred again when terlipressin therapy was reattempted.</p><p><strong>Conclusion: </strong>Vasopressin is known to sensitize baroreceptor reflexes by a central mechanism though its actions on V1a receptors in the area postrema, and we speculate that vasopressin analogues such as terlipressin may act in the same manner. That this effect is not widely described in terlipressin safety literature may be due to the overall younger age range of the trial population. This raises the possibility that cardiac monitoring may be warranted for elderly patients receiving terlipressin.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"340-346"},"PeriodicalIF":0.5,"publicationDate":"2024-06-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250250/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141629690","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: Colon cancer has seen a steady decline in incidence due to increased colonoscopy use. We can assume that this increased use, results in a higher incidence of post-colonoscopy complications such postpolypectomy syndrome, perforation and post-colonoscopy appendicitis (PCA). In this report, we present a case of PCA presenting to the emergency department within 12 h of a screening colonoscopy.
Case presentation: Our patient, a 77-year-old male, underwent an uncomplicated screening colonoscopy and was discharged home after briefly being monitored without any complaints. Later that day, the patient presented to the emergency department complaining of acute generalized abdominal pain. On presentation, the patient was found to be hypertensive and febrile with a distended abdomen with right lower quadrant tenderness on examination. Laboratory investigations noted an elevated white blood cell count with no evidence of acute appendicitis or focal inflammatory changes on contrast-enhanced abdominal and pelvic computer tomography. The patient was subsequently admitted and developed worsening right lower quadrant abdominal pain and distention overnight. Due to this worsening clinical condition, the decision was made to proceed with a diagnostic laparoscopy. After frank pus was found laparoscopically around the cecum and appendix, it was then converted to an exploratory laparotomy. Subsequently, a perforated gangrenous appendix was found with an erythematous and indurated cecum.
Conclusion: Major complications of colonoscopy can include perforation and/or post-colonoscopy bleeding which have been shown to have a respective incidence of 0.21% and 0.1%. With the anticipated rise in the number of colonoscopies, much rarer complications such as PCA with an incidence of less than 0.05% will be seen more frequently. Due to its nonspecific presentation, it is necessary for providers to consider PCA as an important differential for all patients presenting with abdominal pain after a colonoscopy.
{"title":"A Rapid Development of Post-Colonoscopy Appendicitis within Twelve Hours: A Case Report.","authors":"Sean-Patrick Prince, Qitan Huang, Denisse Camille Dayto, Andrew Sephien, Varun Patel, Sreekanth Chandrupatla","doi":"10.1159/000538970","DOIUrl":"10.1159/000538970","url":null,"abstract":"<p><strong>Introduction: </strong>Colon cancer has seen a steady decline in incidence due to increased colonoscopy use. We can assume that this increased use, results in a higher incidence of post-colonoscopy complications such postpolypectomy syndrome, perforation and post-colonoscopy appendicitis (PCA). In this report, we present a case of PCA presenting to the emergency department within 12 h of a screening colonoscopy.</p><p><strong>Case presentation: </strong>Our patient, a 77-year-old male, underwent an uncomplicated screening colonoscopy and was discharged home after briefly being monitored without any complaints. Later that day, the patient presented to the emergency department complaining of acute generalized abdominal pain. On presentation, the patient was found to be hypertensive and febrile with a distended abdomen with right lower quadrant tenderness on examination. Laboratory investigations noted an elevated white blood cell count with no evidence of acute appendicitis or focal inflammatory changes on contrast-enhanced abdominal and pelvic computer tomography. The patient was subsequently admitted and developed worsening right lower quadrant abdominal pain and distention overnight. Due to this worsening clinical condition, the decision was made to proceed with a diagnostic laparoscopy. After frank pus was found laparoscopically around the cecum and appendix, it was then converted to an exploratory laparotomy. Subsequently, a perforated gangrenous appendix was found with an erythematous and indurated cecum.</p><p><strong>Conclusion: </strong>Major complications of colonoscopy can include perforation and/or post-colonoscopy bleeding which have been shown to have a respective incidence of 0.21% and 0.1%. With the anticipated rise in the number of colonoscopies, much rarer complications such as PCA with an incidence of less than 0.05% will be seen more frequently. Due to its nonspecific presentation, it is necessary for providers to consider PCA as an important differential for all patients presenting with abdominal pain after a colonoscopy.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"333-339"},"PeriodicalIF":0.5,"publicationDate":"2024-06-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11249547/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141629688","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: Anisakiasis is a parasitic disease caused by larvae from anisakid nematodes. In recent years, there has been an increase in cases of anisakiasis, a relatively uncommon medical condition.
Case presentation: A 93-year-old woman with chronic heart failure developed hives and nausea 6 h after eating silver flounder sashimi, leading to suspicion of fish allergy. Despite treatment, symptoms persisted and progressed to abdominal pain. An upper gastrointestinal endoscopy revealed an Anisakis larva in her stomach, an unusual finding given her age. After endoscopic removal of the living worm, the patient's symptoms were relieved.
Conclusion: A case of atypical anisakiasis in a 93-year-old patient was demonstrated. This case underscores the importance of considering atypical presentations in clinical decision-making.
{"title":"A Case of Nonagenarian Gastric Anisakiasis with Atypical Presentation.","authors":"Toshio Arai, Seisuke Saito, Takahiro Kinebuchi, Koji Nishikawa, Yasuyuki Morishima, Hiromu Sugiyama","doi":"10.1159/000539304","DOIUrl":"10.1159/000539304","url":null,"abstract":"<p><strong>Introduction: </strong>Anisakiasis is a parasitic disease caused by larvae from anisakid nematodes. In recent years, there has been an increase in cases of anisakiasis, a relatively uncommon medical condition.</p><p><strong>Case presentation: </strong>A 93-year-old woman with chronic heart failure developed hives and nausea 6 h after eating silver flounder sashimi, leading to suspicion of fish allergy. Despite treatment, symptoms persisted and progressed to abdominal pain. An upper gastrointestinal endoscopy revealed an <i>Anisakis</i> larva in her stomach, an unusual finding given her age. After endoscopic removal of the living worm, the patient's symptoms were relieved.</p><p><strong>Conclusion: </strong>A case of atypical anisakiasis in a 93-year-old patient was demonstrated. This case underscores the importance of considering atypical presentations in clinical decision-making.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"327-332"},"PeriodicalIF":0.5,"publicationDate":"2024-06-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250682/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626096","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 : 2024-06-14eCollection Date: 2024-01-01DOI: 10.1159/000539413
Akiyoshi Tsuboi, Shuya Shigenobu, Yuka Matsubara, Issei Hirata, Hidenori Tanaka, Ken Yamashita, Ryo Yuge, Yuji Urabe, Koji Arihiro, Shiro Oka
Introduction: Coronavirus disease 2019 (COVID-19) caused by the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is frequently associated with various gastrointestinal symptoms, including abdominal pain, vomiting, and diarrhea. Moreover, several cases of refractory diarrhea have been reported after COVID-19 recovery. Herein, we present a case of severe refractory diarrhea associated with COVID-19.
Case presentation: A 50-year-old man with no comorbidities was admitted to our hospital with SARS-CoV-2 pneumonia. His respiratory status deteriorated, and ventilatory management, including extracorporeal membrane oxygenation, was needed. The patient's respiratory condition improved, resulting in a transfer to another hospital for rehabilitation. However, the patient developed diarrhea that worsened to 6,000-7,000 mL/day, and he was transferred to our hospital. We diagnosed the patient with enterocolitis caused by cytomegalovirus infection and treated him with ganciclovir on day 5 after transfer to our hospital. The diarrhea did not improve. We suspected enterocolitis associated with COVID-19 and administered a methylprednisolone pulse (intravenous injection, 1,000 mg/day for 3 days) on day 10 after transfer, resulting in a marked improvement in his symptoms. The prednisolone dose was tapered, and no recurrence of diarrhea was observed thereafter.
Conclusion: The prevalence of COVID-19-associated enterocolitis is low, and the pathogenesis of the disease remains unclear. Prednisolone administration should be considered in cases of post-COVID-19 symptoms of severe diarrhea due to a possible abnormal immune response related to COVID-19.
{"title":"Severe Refractory Diarrhea Associated with COVID-19: A Case Report.","authors":"Akiyoshi Tsuboi, Shuya Shigenobu, Yuka Matsubara, Issei Hirata, Hidenori Tanaka, Ken Yamashita, Ryo Yuge, Yuji Urabe, Koji Arihiro, Shiro Oka","doi":"10.1159/000539413","DOIUrl":"10.1159/000539413","url":null,"abstract":"<p><strong>Introduction: </strong>Coronavirus disease 2019 (COVID-19) caused by the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is frequently associated with various gastrointestinal symptoms, including abdominal pain, vomiting, and diarrhea. Moreover, several cases of refractory diarrhea have been reported after COVID-19 recovery. Herein, we present a case of severe refractory diarrhea associated with COVID-19.</p><p><strong>Case presentation: </strong>A 50-year-old man with no comorbidities was admitted to our hospital with SARS-CoV-2 pneumonia. His respiratory status deteriorated, and ventilatory management, including extracorporeal membrane oxygenation, was needed. The patient's respiratory condition improved, resulting in a transfer to another hospital for rehabilitation. However, the patient developed diarrhea that worsened to 6,000-7,000 mL/day, and he was transferred to our hospital. We diagnosed the patient with enterocolitis caused by cytomegalovirus infection and treated him with ganciclovir on day 5 after transfer to our hospital. The diarrhea did not improve. We suspected enterocolitis associated with COVID-19 and administered a methylprednisolone pulse (intravenous injection, 1,000 mg/day for 3 days) on day 10 after transfer, resulting in a marked improvement in his symptoms. The prednisolone dose was tapered, and no recurrence of diarrhea was observed thereafter.</p><p><strong>Conclusion: </strong>The prevalence of COVID-19-associated enterocolitis is low, and the pathogenesis of the disease remains unclear. Prednisolone administration should be considered in cases of post-COVID-19 symptoms of severe diarrhea due to a possible abnormal immune response related to COVID-19.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"318-326"},"PeriodicalIF":0.5,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250576/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626100","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 : 2024-06-13eCollection Date: 2024-01-01DOI: 10.1159/000539288
Rahil Desai, Katherine Khazey, Hasnoor Sandhu, Peter Makar, Navkiran Randhawa, Ahamed Khalyfa, Mahnoor Khan, Alex Yarbrough, Tilemahos Spyratos
Introduction: Mantle cell lymphoma (MCL), a rare non-Hodgkin's lymphoma, exhibits a genetic translocation causing CCND1 gene overexpression, affecting 5% of NHL cases, predominantly in males aged 60-70. Typically diagnosed with advanced symptoms, MCL involves widespread disease and organ spread, being aggressive and incurable with a 1.8-9.4-year average survival. Optimal treatment depends on disease aggressiveness and age. Multiple lymphomatous polyposis (MLP), a rare MCL subtype in the GI tract, is usually present with GI symptoms.
Case presentation: A 71-year-old woman was diagnosed with asymptomatic MLP during MCL staging who underwent thoracentesis due to large right pleural effusion and significant axillary lymphadenopathy and was treated with a chemotherapy regimen of rituximab/cytarabine and later transitioned to bendamustine/rituximab. This patient eventually underwent a bone marrow biopsy and later a bone marrow transplant.
Conclusion: We present a unique case of asymptomatic MLP, emphasizing the importance of early detection for the poor prognosis of MLP with a mean survival of less than 3 years.
{"title":"Gastrointestinal Symptom-Free Multiple Lymphomatous Polyposis: An Atypical Case Presentation of Mantle Cell Lymphoma.","authors":"Rahil Desai, Katherine Khazey, Hasnoor Sandhu, Peter Makar, Navkiran Randhawa, Ahamed Khalyfa, Mahnoor Khan, Alex Yarbrough, Tilemahos Spyratos","doi":"10.1159/000539288","DOIUrl":"10.1159/000539288","url":null,"abstract":"<p><strong>Introduction: </strong>Mantle cell lymphoma (MCL), a rare non-Hodgkin's lymphoma, exhibits a genetic translocation causing CCND1 gene overexpression, affecting 5% of NHL cases, predominantly in males aged 60-70. Typically diagnosed with advanced symptoms, MCL involves widespread disease and organ spread, being aggressive and incurable with a 1.8-9.4-year average survival. Optimal treatment depends on disease aggressiveness and age. Multiple lymphomatous polyposis (MLP), a rare MCL subtype in the GI tract, is usually present with GI symptoms.</p><p><strong>Case presentation: </strong>A 71-year-old woman was diagnosed with asymptomatic MLP during MCL staging who underwent thoracentesis due to large right pleural effusion and significant axillary lymphadenopathy and was treated with a chemotherapy regimen of rituximab/cytarabine and later transitioned to bendamustine/rituximab. This patient eventually underwent a bone marrow biopsy and later a bone marrow transplant.</p><p><strong>Conclusion: </strong>We present a unique case of asymptomatic MLP, emphasizing the importance of early detection for the poor prognosis of MLP with a mean survival of less than 3 years.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"313-317"},"PeriodicalIF":0.5,"publicationDate":"2024-06-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250683/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141629689","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 : 2024-06-07eCollection Date: 2024-01-01DOI: 10.1159/000538688
Raymond Fueng-Hin Liang, Cora Yuk-Ping Chau, Wee Chian Lim
Introduction: Gastrointestinal stromal tumours (GISTs) are an important, though uncommon, cause of obscure gastrointestinal bleeding and may rarely be associated with genodermatoses such as neurofibromatosis type 1 (NF1). NF1-related GISTs have unique phenotypic features compared with sporadic GISTs and may elude diagnosis due to their predilection for the small bowel.
Case presentation: We report a case of a 45-year-old Singaporean woman with café-au-lait macules and cutaneous neurofibromas who presented with occult obscure gastrointestinal bleeding and was eventually discovered to have a bleeding jejunal GIST. This finding, considered together with her cutaneous signs, eventually led to the diagnosis of NF1.
Conclusion: Genodermatoses and their gastrointestinal complications are likely under-reported in adult Southeast Asian populations and deserve greater awareness from gastroenterologists practising in this region.
{"title":"Neurofibromatosis Type 1 Presenting as Bleeding Jejunal Gastrointestinal Stromal Tumour.","authors":"Raymond Fueng-Hin Liang, Cora Yuk-Ping Chau, Wee Chian Lim","doi":"10.1159/000538688","DOIUrl":"10.1159/000538688","url":null,"abstract":"<p><strong>Introduction: </strong>Gastrointestinal stromal tumours (GISTs) are an important, though uncommon, cause of obscure gastrointestinal bleeding and may rarely be associated with genodermatoses such as neurofibromatosis type 1 (NF1). NF1-related GISTs have unique phenotypic features compared with sporadic GISTs and may elude diagnosis due to their predilection for the small bowel.</p><p><strong>Case presentation: </strong>We report a case of a 45-year-old Singaporean woman with café-au-lait macules and cutaneous neurofibromas who presented with occult obscure gastrointestinal bleeding and was eventually discovered to have a bleeding jejunal GIST. This finding, considered together with her cutaneous signs, eventually led to the diagnosis of NF1.</p><p><strong>Conclusion: </strong>Genodermatoses and their gastrointestinal complications are likely under-reported in adult Southeast Asian populations and deserve greater awareness from gastroenterologists practising in this region.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"299-305"},"PeriodicalIF":0.6,"publicationDate":"2024-06-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11185854/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141417906","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 : 2024-06-07eCollection Date: 2024-01-01DOI: 10.1159/000538965
Simone A Jarrett, Rushi Kaushik Talati, Johann Hasbun, Wenqing Cao, Scott M Smukalla
Introduction: Acute upper gastrointestinal bleeding is one of the most common medical emergencies that present to the hospital, and delineating the underlying etiology is essential to provide adequate definitive treatment. The purpose of this case report was to review the diagnosis and treatment of a rare complication known as radiation-induced hemorrhagic gastritis (RIHG) that can occur in patients with prior radiation exposure. The motivation for this study arose from the identification of a case within our institution.
Case presentation: The study involved a review of the diagnosis and management of a patient who presented with anemia and recurrent episodes of gastrointestinal bleeding at our institution after undergoing treatment for metastatic biliary adenocarcinoma. With the advent of new therapies, we aimed to investigate the various techniques utilized to manage these patients and highlight the importance of maintaining a high index of suspicion for RIHG as a potential etiology of gastrointestinal bleeding in patients with a relevant medical history of radiation exposure. Despite the literature review, we found that there is a lack of guidelines in the approach to the management of these patients.
Conclusion: This case report underscores the rarity of radiation-induced gastritis and the complications that may arise from its diagnosis, including recurrent GI bleeding. Further investigation into identifying definitive treatment and creating guidelines for its management is desperately needed.
{"title":"Recurrent Upper Gastrointestinal Bleeding due to Radiation-Induced Hemorrhagic Gastroduodenal Ectasia: A Review of Current Treatment Options for Radiation-Induced Gastric Injury.","authors":"Simone A Jarrett, Rushi Kaushik Talati, Johann Hasbun, Wenqing Cao, Scott M Smukalla","doi":"10.1159/000538965","DOIUrl":"10.1159/000538965","url":null,"abstract":"<p><strong>Introduction: </strong>Acute upper gastrointestinal bleeding is one of the most common medical emergencies that present to the hospital, and delineating the underlying etiology is essential to provide adequate definitive treatment. The purpose of this case report was to review the diagnosis and treatment of a rare complication known as radiation-induced hemorrhagic gastritis (RIHG) that can occur in patients with prior radiation exposure. The motivation for this study arose from the identification of a case within our institution.</p><p><strong>Case presentation: </strong>The study involved a review of the diagnosis and management of a patient who presented with anemia and recurrent episodes of gastrointestinal bleeding at our institution after undergoing treatment for metastatic biliary adenocarcinoma. With the advent of new therapies, we aimed to investigate the various techniques utilized to manage these patients and highlight the importance of maintaining a high index of suspicion for RIHG as a potential etiology of gastrointestinal bleeding in patients with a relevant medical history of radiation exposure. Despite the literature review, we found that there is a lack of guidelines in the approach to the management of these patients.</p><p><strong>Conclusion: </strong>This case report underscores the rarity of radiation-induced gastritis and the complications that may arise from its diagnosis, including recurrent GI bleeding. Further investigation into identifying definitive treatment and creating guidelines for its management is desperately needed.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"306-312"},"PeriodicalIF":0.5,"publicationDate":"2024-06-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11249716/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626099","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 : 2024-05-31eCollection Date: 2024-01-01DOI: 10.1159/000538794
Shubhang K Bhatt, Nikita Ashcherkin, John Fanous, Rish K Pai, Janhavi Athale
Introduction: Langerhans cell histiocytosis (LCH) is a rare hematologic condition which can affect multiple organ systems and has variable presentation. LCH is more commonly seen as a malignancy of childhood. LCH in adulthood can have poor outcomes depending on the involvement of critical organs.
Case presentation: We report a case of a 71-year-old female who presented with progressive weakness, weight loss, diarrhea, and jaundice, and had been undergoing outpatient workup for elevated liver enzymes for the last 2 years. She required admission to the intensive care unit for vasodilatory shock, requiring vasopressor and chronotropic support. Imaging showed an underlying multiorgan process involving the gastrointestinal tract, liver, spleen, and central nervous system. A repeat liver biopsy after a prior inconclusive one revealed the diagnosis of multisystem LCH presenting as secondary sclerosing cholangitis.
Conclusion: The uniqueness of this multisystem LCH case lies not only in its rarity but also in the diagnostic journey that necessitated a repeat biopsy for a conclusive diagnosis. Early identification and targeted intervention can help in ensuring better patient outcomes, especially when the presentation can overlap with various other possible conditions.
{"title":"Hepatitis and Vasodilatory Shock due to an Unsuspected Culprit: A Rare Presentation of Multisystem Langerhans Cell Histiocytosis.","authors":"Shubhang K Bhatt, Nikita Ashcherkin, John Fanous, Rish K Pai, Janhavi Athale","doi":"10.1159/000538794","DOIUrl":"10.1159/000538794","url":null,"abstract":"<p><strong>Introduction: </strong>Langerhans cell histiocytosis (LCH) is a rare hematologic condition which can affect multiple organ systems and has variable presentation. LCH is more commonly seen as a malignancy of childhood. LCH in adulthood can have poor outcomes depending on the involvement of critical organs.</p><p><strong>Case presentation: </strong>We report a case of a 71-year-old female who presented with progressive weakness, weight loss, diarrhea, and jaundice, and had been undergoing outpatient workup for elevated liver enzymes for the last 2 years. She required admission to the intensive care unit for vasodilatory shock, requiring vasopressor and chronotropic support. Imaging showed an underlying multiorgan process involving the gastrointestinal tract, liver, spleen, and central nervous system. A repeat liver biopsy after a prior inconclusive one revealed the diagnosis of multisystem LCH presenting as secondary sclerosing cholangitis.</p><p><strong>Conclusion: </strong>The uniqueness of this multisystem LCH case lies not only in its rarity but also in the diagnostic journey that necessitated a repeat biopsy for a conclusive diagnosis. Early identification and targeted intervention can help in ensuring better patient outcomes, especially when the presentation can overlap with various other possible conditions.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"286-292"},"PeriodicalIF":0.6,"publicationDate":"2024-05-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11167066/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141310138","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 : 2024-05-31eCollection Date: 2024-01-01DOI: 10.1159/000538833
Yuji Urabe, Hideki Ishikawa, Akira Ishikawa, Shingo Ishiguro, Kazuki Ishibashi, Koji Arihiro, Michihiro Mutoh, Shiro Oka
Introduction: Familial adenomatous polyposis (FAP), a hereditary disorder of the gastrointestinal tract, is an autosomal dominant inherited condition caused by germline mutations in the adenomatous polyposis coli (APC) gene. It is characterized by the development of hundreds to thousands of colorectal adenomatous polyps, which, if left untreated, can eventually develop into colorectal carcinomas. Representative extracolonic tumors in FAP include multiple duodenal adenomas and desmoid tumors. Moreover, multiple fundic gland polyps are frequently identified in the stomachs of patients with FAP.
Case presentation: Herein, we report the two cases. A 52-year-old woman who underwent total colectomy for FAP, and pancreatoduodenectomy was initiated on esomeprazole for the treatment of anastomotic erosion. Esophagogastroduodenoscopy performed 42 months later showed an increased number and size of gastric fundic gland polyps, which subsequently decreased after replacing esomeprazole with ranitidine. Similarly, a 39-year-old woman with FAP was initiated on vonoprazan for the treatment of reflux symptoms. Esophagogastroduodenoscopy and colonoscopy performed 14 months later indicated an increase in the number of gastric fundic gland polyps and colorectal polyps, which subsequently decreased after vonoprazan discontinuation. In these two cases, the increase and decrease in the number and size of fundic gland polyps and colon adenoma were associated with serum gastrin levels.
Conclusion: Gastric fundic gland polyps and colon polyps may rapidly increase in number and size due to increased gastrin levels induced by proton pump inhibitor/potassium-competitive acid blocker use. Hence, these drugs should be prescribed with caution.
{"title":"Two Cases of Increased Gastrointestinal Polyps in Familial Adenomatous Polyposis following Antiacid Agent Intake.","authors":"Yuji Urabe, Hideki Ishikawa, Akira Ishikawa, Shingo Ishiguro, Kazuki Ishibashi, Koji Arihiro, Michihiro Mutoh, Shiro Oka","doi":"10.1159/000538833","DOIUrl":"10.1159/000538833","url":null,"abstract":"<p><strong>Introduction: </strong>Familial adenomatous polyposis (FAP), a hereditary disorder of the gastrointestinal tract, is an autosomal dominant inherited condition caused by germline mutations in the adenomatous polyposis coli (<i>APC</i>) gene. It is characterized by the development of hundreds to thousands of colorectal adenomatous polyps, which, if left untreated, can eventually develop into colorectal carcinomas. Representative extracolonic tumors in FAP include multiple duodenal adenomas and desmoid tumors. Moreover, multiple fundic gland polyps are frequently identified in the stomachs of patients with FAP.</p><p><strong>Case presentation: </strong>Herein, we report the two cases. A 52-year-old woman who underwent total colectomy for FAP, and pancreatoduodenectomy was initiated on esomeprazole for the treatment of anastomotic erosion. Esophagogastroduodenoscopy performed 42 months later showed an increased number and size of gastric fundic gland polyps, which subsequently decreased after replacing esomeprazole with ranitidine. Similarly, a 39-year-old woman with FAP was initiated on vonoprazan for the treatment of reflux symptoms. Esophagogastroduodenoscopy and colonoscopy performed 14 months later indicated an increase in the number of gastric fundic gland polyps and colorectal polyps, which subsequently decreased after vonoprazan discontinuation. In these two cases, the increase and decrease in the number and size of fundic gland polyps and colon adenoma were associated with serum gastrin levels.</p><p><strong>Conclusion: </strong>Gastric fundic gland polyps and colon polyps may rapidly increase in number and size due to increased gastrin levels induced by proton pump inhibitor/potassium-competitive acid blocker use. Hence, these drugs should be prescribed with caution.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"293-298"},"PeriodicalIF":0.5,"publicationDate":"2024-05-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250236/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626101","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}