Introduction: Hemobilia, which refers to bleeding from the bile duct, is rare and difficult to treat. We report a case of successful hemostasis of a pancreatic tumor complicated by hemobilia.
Case presentation: A 76-year-old man was referred to our hospital with a pancreatic head tumor. Endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic ultrasonography-FNA were performed, and the patient was diagnosed with pancreatic metastasis of renal cell carcinoma. After discharge, the patient noted worsening jaundice and progressive anemia and was readmitted. ERCP reveals active bleeding from the duodenal papillae. The patient was placed on a fully covered metallic stent and discharged after confirming hemostasis.
Conclusion: Renal cell carcinoma is a tumor with abundant blood flow. If hemobilia occurs, bleeding from pancreatic metastatic tumors should be considered. Additionally, hemostasis using a fully covered metallic stent is useful for treating hemobilia in tumors.
{"title":"Hemostasis with Metallic Stent for Multiple Metastatic Pancreatic Tumors Complicated with Hemobilia.","authors":"Hiroyuki Ito, Yuji Omura, Tomonori Makuuchi, Tsubomi Chou, Ayano Ito, Ryutaro Fujimoto, Masashi Yokota, Shingo Tsuda, Junko Nagata, Shunji Hirose, Hisamichi Yoshii, Hideki Izumi, Tomoko Sugiyama, Takuma Tajiri, Takayoshi Suzuki","doi":"10.1159/000536221","DOIUrl":"10.1159/000536221","url":null,"abstract":"<p><strong>Introduction: </strong>Hemobilia, which refers to bleeding from the bile duct, is rare and difficult to treat. We report a case of successful hemostasis of a pancreatic tumor complicated by hemobilia.</p><p><strong>Case presentation: </strong>A 76-year-old man was referred to our hospital with a pancreatic head tumor. Endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic ultrasonography-FNA were performed, and the patient was diagnosed with pancreatic metastasis of renal cell carcinoma. After discharge, the patient noted worsening jaundice and progressive anemia and was readmitted. ERCP reveals active bleeding from the duodenal papillae. The patient was placed on a fully covered metallic stent and discharged after confirming hemostasis.</p><p><strong>Conclusion: </strong>Renal cell carcinoma is a tumor with abundant blood flow. If hemobilia occurs, bleeding from pancreatic metastatic tumors should be considered. Additionally, hemostasis using a fully covered metallic stent is useful for treating hemobilia in tumors.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-02-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10846876/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139696942","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 : 2024-02-01eCollection Date: 2024-01-01DOI: 10.1159/000536117
Harish Gopalakrishna, Bilal Asif, Anjali Rai, Hari S Conjeevaram, Maria Mironova, David E Kleiner, Alexandra F Freeman, Theo Heller
Introduction: Prolidase deficiency is a rare autosomal recessive disorder caused by variants in the PEPD gene. Patients usually have multi-organ involvement and a wide range of clinical features including recurrent skin ulcers, dysmorphic facial features, recurrent infections, intellectual disability, and splenomegaly. Studies have shown that patients with prolidase deficiency may have hepatic manifestations including hepatomegaly and abnormal liver enzymes. However, there is no detailed description of liver disease in this patient population.
Case presentation: Here, we present 3 patients with prolidase deficiency with varying extents of hepatic involvement.
Conclusion: Prolidase deficiency patients with liver disease should be followed up long term to understand more about the pathophysiology and the impact of liver disease on long-term outcomes.
{"title":"Chronic Liver Disease in Patients with Prolidase Deficiency: A Case Series.","authors":"Harish Gopalakrishna, Bilal Asif, Anjali Rai, Hari S Conjeevaram, Maria Mironova, David E Kleiner, Alexandra F Freeman, Theo Heller","doi":"10.1159/000536117","DOIUrl":"https://doi.org/10.1159/000536117","url":null,"abstract":"<p><strong>Introduction: </strong>Prolidase deficiency is a rare autosomal recessive disorder caused by variants in the <i>PEPD</i> gene. Patients usually have multi-organ involvement and a wide range of clinical features including recurrent skin ulcers, dysmorphic facial features, recurrent infections, intellectual disability, and splenomegaly. Studies have shown that patients with prolidase deficiency may have hepatic manifestations including hepatomegaly and abnormal liver enzymes. However, there is no detailed description of liver disease in this patient population.</p><p><strong>Case presentation: </strong>Here, we present 3 patients with prolidase deficiency with varying extents of hepatic involvement.</p><p><strong>Conclusion: </strong>Prolidase deficiency patients with liver disease should be followed up long term to understand more about the pathophysiology and the impact of liver disease on long-term outcomes.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10834036/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139671378","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 : 2024-01-29eCollection Date: 2024-01-01DOI: 10.1159/000536097
Asuka Watanabe, Tsutomu Nishida, Naoto Osugi, Takao Kitanaka, Yutaro Minoura, Satoru Okabe, Naohiro Sakamoto, Yoshifumi Fujii, Aya Sugimoto, Dai Nakamatsu, Kengo Matsumoto, Masashi Yamamoto, Shiro Adachi, Koji Fukui
Introduction: Drug-induced liver injury (DILI) associated with 5-aminosalicylic acid (5-ASA) is a rare but potentially life-threatening adverse event.
Case presentation: We report the case of a 58-year-old woman with ulcerative colitis who developed DILI after initiating maintenance therapy with the multimatrix system 5-ASA. The patient presented with grade 4 liver enzyme elevation on day 98 after initiating 5-ASA and was admitted to the hospital. Blood tests revealed the mixed liver injury, and imaging studies showed no abnormalities except for mild lymph node enlargement. Liver biopsy revealed acute lobular hepatitis with interfacial activity. The patient's score on the International Autoimmune Hepatitis Group 1999 revised scoring system was a total score of 10, causing a suspicion for the diagnosis of autoimmune hepatitis. The DDW-J 2004 scale calculated a total score of six, indicating a high probability of DILI. We suspected DILI due to 5-ASA, and the 5-ASA formulations were discontinued. The patient was treated with ursodeoxycholic acid and neominophagen C, and her liver function gradually improved without steroid treatment. Finally, we definitively diagnosed DILI based on the pathological findings and clinical course after discontinuation of 5-ASA.
Conclusion: This case highlights the importance of monitoring liver function in patients receiving 5-ASA therapy.
{"title":"5-Aminosalicylic Acid-Induced Liver Injury in a Patient with Ulcerative Colitis: A Case Report.","authors":"Asuka Watanabe, Tsutomu Nishida, Naoto Osugi, Takao Kitanaka, Yutaro Minoura, Satoru Okabe, Naohiro Sakamoto, Yoshifumi Fujii, Aya Sugimoto, Dai Nakamatsu, Kengo Matsumoto, Masashi Yamamoto, Shiro Adachi, Koji Fukui","doi":"10.1159/000536097","DOIUrl":"10.1159/000536097","url":null,"abstract":"<p><strong>Introduction: </strong>Drug-induced liver injury (DILI) associated with 5-aminosalicylic acid (5-ASA) is a rare but potentially life-threatening adverse event.</p><p><strong>Case presentation: </strong>We report the case of a 58-year-old woman with ulcerative colitis who developed DILI after initiating maintenance therapy with the multimatrix system 5-ASA. The patient presented with grade 4 liver enzyme elevation on day 98 after initiating 5-ASA and was admitted to the hospital. Blood tests revealed the mixed liver injury, and imaging studies showed no abnormalities except for mild lymph node enlargement. Liver biopsy revealed acute lobular hepatitis with interfacial activity. The patient's score on the International Autoimmune Hepatitis Group 1999 revised scoring system was a total score of 10, causing a suspicion for the diagnosis of autoimmune hepatitis. The DDW-J 2004 scale calculated a total score of six, indicating a high probability of DILI. We suspected DILI due to 5-ASA, and the 5-ASA formulations were discontinued. The patient was treated with ursodeoxycholic acid and neominophagen C, and her liver function gradually improved without steroid treatment. Finally, we definitively diagnosed DILI based on the pathological findings and clinical course after discontinuation of 5-ASA.</p><p><strong>Conclusion: </strong>This case highlights the importance of monitoring liver function in patients receiving 5-ASA therapy.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-01-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10824519/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139575261","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 : 2024-01-19eCollection Date: 2024-01-01DOI: 10.1159/000535988
Kaoru Abe, Daisuke Yamai, Chihiro Katsumi, Manabu Oyamatsu, Kenji Sato
Introduction: A stoma prolapse is easy to diagnose by visual examination, and it rarely incarcerates. Therefore, manual reduction is usually performed as soon as the diagnosis is made. In this report, we describe a case of stoma prolapse that could not be reduced manually and ruptured because an incarcerated parastomal hernia occurred in the stoma, mimicking stoma prolapse.
Case presentation: A 66-year-old woman underwent total hysterectomy, bilateral salpingo-oophorectomy, pelvic and para-aortic lymphadenectomy, omentectomy, resection of dissemination, and low anterior resection with formation of a sigmoid end colostomy for endometrial cancer with infiltration of the rectum. Fourteen months after the initial operation, she presented with stoma prolapse and multiple episodes of vomiting. The prolapsed stoma was 20 cm in length, appeared swollen and edematous, and was somewhat firm. Although it looked viable, some of the mucosa was darkish red, indicating congestion. Therefore, the diagnosis was sigmoid end colostomy prolapse with an ischemic component. An attempt at manual reduction resulted in rupture, so an emergency laparotomy was performed. Intraoperatively, we found that the ileum was incarcerated in the aperture created where the colostomy had been formed. When the incarcerated ileum was released, the stoma prolapse could be reduced easily. The end colostomy was refashioned in the left upper quadrant of the abdomen.
Conclusion: An incarcerated parastomal hernia can mimic stoma prolapse. If the findings differ from those of typical stoma prolapse, imaging should be performed to confirm whether another clinical entity is involved in the stoma prolapse.
{"title":"Incarcerated Small Bowel Herniation in a Stoma Mimicking Sigmoid End Colostomy Prolapse.","authors":"Kaoru Abe, Daisuke Yamai, Chihiro Katsumi, Manabu Oyamatsu, Kenji Sato","doi":"10.1159/000535988","DOIUrl":"10.1159/000535988","url":null,"abstract":"<p><strong>Introduction: </strong>A stoma prolapse is easy to diagnose by visual examination, and it rarely incarcerates. Therefore, manual reduction is usually performed as soon as the diagnosis is made. In this report, we describe a case of stoma prolapse that could not be reduced manually and ruptured because an incarcerated parastomal hernia occurred in the stoma, mimicking stoma prolapse.</p><p><strong>Case presentation: </strong>A 66-year-old woman underwent total hysterectomy, bilateral salpingo-oophorectomy, pelvic and para-aortic lymphadenectomy, omentectomy, resection of dissemination, and low anterior resection with formation of a sigmoid end colostomy for endometrial cancer with infiltration of the rectum. Fourteen months after the initial operation, she presented with stoma prolapse and multiple episodes of vomiting. The prolapsed stoma was 20 cm in length, appeared swollen and edematous, and was somewhat firm. Although it looked viable, some of the mucosa was darkish red, indicating congestion. Therefore, the diagnosis was sigmoid end colostomy prolapse with an ischemic component. An attempt at manual reduction resulted in rupture, so an emergency laparotomy was performed. Intraoperatively, we found that the ileum was incarcerated in the aperture created where the colostomy had been formed. When the incarcerated ileum was released, the stoma prolapse could be reduced easily. The end colostomy was refashioned in the left upper quadrant of the abdomen.</p><p><strong>Conclusion: </strong>An incarcerated parastomal hernia can mimic stoma prolapse. If the findings differ from those of typical stoma prolapse, imaging should be performed to confirm whether another clinical entity is involved in the stoma prolapse.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-01-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10798682/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139511897","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 : 2024-01-19eCollection Date: 2024-01-01DOI: 10.1159/000534548
Kinga Szopińska, Karolina Tracz, Żaneta Malczyk, Anna Jarzumbek, Andrzej Grabowski, Katarzyna Bąk-Drabik
Introduction: Bezoars are masses of indigestible foreign material in the gastrointestinal tract, usually in the stomach. These materials could be indigestible fruits, hair, milk products, or tablets. In children, the most common type of bezoar is trichobezoar (formed from hair).
Case presentation: We describe a female patient who has been complaining about deterioration of mood, collapse without losing consciousness, scotomas, and cardiac arrhythmia for 2 years. Based on the results of thyroid hormone, resistance to thyroid hormone (RTH) was suspected. Physical examination during hospitalization revealed a palpable upper abdominal mass. Several diagnostic examinations were performed. The abdominal ultrasound showed acoustic shadowing caused by a pathological structure in the upper abdomen. Therefore, the contrast X-ray of the digestive tract revealed a deficit of contrast with an irregular shape in the stomach body and the pylorus region. Due to these results, a gastroscopy was performed, which revealed a large trichobezoar of the stomach. The trichobezoar was surgically removed without complications.
Conclusion: The case presented shows that these nonspecific symptoms and laboratory test suggesting RTH require multi-path diagnostics and the cooperation of many specialists, ultimately giving a surprising diagnosis. It is crucial to interpret diagnostic examinations with regard to the patient's physical condition. Diagnosis of trichobezoar requires a detailed search of causes to avoid another incident.
简介牛粪是胃肠道中难以消化的异物块,通常位于胃中。这些物质可能是难以消化的水果、头发、奶制品或药片。在儿童中,最常见的是三叶虫(由头发形成):病例介绍:我们描述了一名女性患者,她主诉情绪恶化、昏倒但不失去知觉、视力模糊和心律失常已有两年。根据甲状腺激素检测结果,怀疑患者存在甲状腺激素抵抗(RTH)。住院期间的体格检查发现了一个可触及的上腹部肿块。对其进行了多项诊断性检查。腹部超声波检查显示,上腹部的病理结构引起了声影。因此,消化道造影剂 X 光检查显示胃体和幽门区域造影剂不足,形状不规则。根据上述结果,医生进行了胃镜检查,发现胃部有一个巨大的三叶虫。手术切除了该三叶虫,未出现并发症:本病例表明,这些非特异性症状和实验室检查提示的 RTH 需要多途径诊断和多位专家的合作,最终才能得出令人惊讶的诊断结果。根据患者的身体状况解释诊断检查至关重要。诊断三叶虫病需要详细查找病因,以避免再次发生。
{"title":"Trichobezoar Found Accidently while Diagnosing Resistance to Thyroid Hormone.","authors":"Kinga Szopińska, Karolina Tracz, Żaneta Malczyk, Anna Jarzumbek, Andrzej Grabowski, Katarzyna Bąk-Drabik","doi":"10.1159/000534548","DOIUrl":"10.1159/000534548","url":null,"abstract":"<p><strong>Introduction: </strong>Bezoars are masses of indigestible foreign material in the gastrointestinal tract, usually in the stomach. These materials could be indigestible fruits, hair, milk products, or tablets. In children, the most common type of bezoar is trichobezoar (formed from hair).</p><p><strong>Case presentation: </strong>We describe a female patient who has been complaining about deterioration of mood, collapse without losing consciousness, scotomas, and cardiac arrhythmia for 2 years. Based on the results of thyroid hormone, resistance to thyroid hormone (RTH) was suspected. Physical examination during hospitalization revealed a palpable upper abdominal mass. Several diagnostic examinations were performed. The abdominal ultrasound showed acoustic shadowing caused by a pathological structure in the upper abdomen. Therefore, the contrast X-ray of the digestive tract revealed a deficit of contrast with an irregular shape in the stomach body and the pylorus region. Due to these results, a gastroscopy was performed, which revealed a large trichobezoar of the stomach. The trichobezoar was surgically removed without complications.</p><p><strong>Conclusion: </strong>The case presented shows that these nonspecific symptoms and laboratory test suggesting RTH require multi-path diagnostics and the cooperation of many specialists, ultimately giving a surprising diagnosis. It is crucial to interpret diagnostic examinations with regard to the patient's physical condition. Diagnosis of trichobezoar requires a detailed search of causes to avoid another incident.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-01-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10798683/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139511928","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 : 2024-01-09eCollection Date: 2024-01-01DOI: 10.1159/000534973
Simone A Jarrett, Sahana Tito, Matthew Chan, Dominic E Jarrett, Kevin B Lo, Richard DePalma
Introduction: The purpose of this case series was to review a rare subset of tumors known as gastric lipomas, which are typically found incidentally. The motivation for this study arose from the identification of 2 cases within our institution in a short period.
Case presentation: The study involved a review of the diagnosis and management of 2 patients presenting with gastric lipomas at our institution after symptoms of gastrointestinal bleeding. With the advent of new radiologic investigations such as computed tomography and magnetic resonance imaging and advances in endoscopy, there are new approaches to identifying and managing these tumors. On further evaluation of the literature, we found that despite the availability of minimally invasive endoscopic techniques such as mucosal resection and submucosal dissection in the setting of large tumors, most patients tend to have to resort to surgical management.
Conclusion: This case series underscores the rarity of gastric lipomas and their often-incidental discovery. Further investigation into endoscopic approaches for managing these tumors is needed, and additionally, there is a need to explore a potential association between gastric lipomas and malignancy, as chronic inflammation of the overlying mucosa may play a significant role.
{"title":"Gastric Lipomas: A Case Series and Review of the Literature.","authors":"Simone A Jarrett, Sahana Tito, Matthew Chan, Dominic E Jarrett, Kevin B Lo, Richard DePalma","doi":"10.1159/000534973","DOIUrl":"10.1159/000534973","url":null,"abstract":"<p><strong>Introduction: </strong>The purpose of this case series was to review a rare subset of tumors known as gastric lipomas, which are typically found incidentally. The motivation for this study arose from the identification of 2 cases within our institution in a short period.</p><p><strong>Case presentation: </strong>The study involved a review of the diagnosis and management of 2 patients presenting with gastric lipomas at our institution after symptoms of gastrointestinal bleeding. With the advent of new radiologic investigations such as computed tomography and magnetic resonance imaging and advances in endoscopy, there are new approaches to identifying and managing these tumors. On further evaluation of the literature, we found that despite the availability of minimally invasive endoscopic techniques such as mucosal resection and submucosal dissection in the setting of large tumors, most patients tend to have to resort to surgical management.</p><p><strong>Conclusion: </strong>This case series underscores the rarity of gastric lipomas and their often-incidental discovery. Further investigation into endoscopic approaches for managing these tumors is needed, and additionally, there is a need to explore a potential association between gastric lipomas and malignancy, as chronic inflammation of the overlying mucosa may play a significant role.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-01-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10776135/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139401943","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: Immune checkpoint inhibitors are sometimes associated with immune-related adverse events during or after treatment. Among these, aseptic meningitis is a rare and serious complication. We report the first case of atezolizumab-induced aseptic meningitis, which occurred during treatment for advanced hepatocellular carcinoma (HCC).
Case presentation: A 74-year-old woman diagnosed with advanced HCC and treated with first-line atezolizumab plus bevacizumab developed anorexia, fatigue, and fever, after three treatment cycles. Cerebrospinal fluid examination showed slightly increased cell count and protein level but no infection or malignancy. Contrast enhancement along the cerebral sulcus was evident in contrast-enhanced magnetic resonance imaging, and the patient was diagnosed with aseptic meningitis associated with atezolizumab. Steroid therapy soon improved her clinical symptoms, and the contrast enhancement along the cerebral sulcus disappeared.
Conclusion: Clinicians should monitor to avoid serious immune-related adverse events, such as aseptic meningitis, in patients during treatment of HCC with immune checkpoint inhibitors and make the diagnosis as soon as possible.
{"title":"A Case of Immune-Related Aseptic Meningitis during Atezolizumab plus Bevacizumab for Hepatocellular Carcinoma.","authors":"Hiroki Kawanaka, Kazuto Tajiri, Nozomu Muraishi, Aiko Murayama, Takamasa Nukui, Ichiro Yasuda","doi":"10.1159/000535476","DOIUrl":"10.1159/000535476","url":null,"abstract":"<p><strong>Introduction: </strong>Immune checkpoint inhibitors are sometimes associated with immune-related adverse events during or after treatment. Among these, aseptic meningitis is a rare and serious complication. We report the first case of atezolizumab-induced aseptic meningitis, which occurred during treatment for advanced hepatocellular carcinoma (HCC).</p><p><strong>Case presentation: </strong>A 74-year-old woman diagnosed with advanced HCC and treated with first-line atezolizumab plus bevacizumab developed anorexia, fatigue, and fever, after three treatment cycles. Cerebrospinal fluid examination showed slightly increased cell count and protein level but no infection or malignancy. Contrast enhancement along the cerebral sulcus was evident in contrast-enhanced magnetic resonance imaging, and the patient was diagnosed with aseptic meningitis associated with atezolizumab. Steroid therapy soon improved her clinical symptoms, and the contrast enhancement along the cerebral sulcus disappeared.</p><p><strong>Conclusion: </strong>Clinicians should monitor to avoid serious immune-related adverse events, such as aseptic meningitis, in patients during treatment of HCC with immune checkpoint inhibitors and make the diagnosis as soon as possible.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-01-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10766420/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139377233","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 : 2024-01-04eCollection Date: 2024-01-01DOI: 10.1159/000535273
Nishigandha Burute, Olexiy Aseyev, Fauzia Hasan, Erika Allard-Ihala, Walid Shahrour, Ahmed Kotb, William Harris, Radu Rozenberg
Introduction: Primary appendiceal carcinoma is rare and comprises up to 1% of all colorectal malignancies. Its invasion into adjacent organs, such as the bladder and rectum, especially as a presenting characteristic, is even less common.
Case presentation: A 75-year-old asymptomatic male tested positive on a screening fecal immunochemical test (FIT). Colonoscopy showed a rectosigmoid tumor and normal appendiceal orifice. Staging MRI surprisingly showed that the cancer was, in fact, of appendiceal origin, coursed posteriorly to invade the rectosigmoid and form adhesions with the urinary bladder. Staging CT did not show metastatic disease. Low anterior resection, en bloc appendectomy, and right hemicolectomy were performed along with cystectomy and ileal conduit. Hematoxylin and eosin stains showed appendiceal adenocarcinoma invading through the appendiceal wall into the rectal muscularis and submucosa. Features of neuroendocrine carcinoma were not identified on immunohistochemistry. This was a colonic type of adenocarcinoma of the appendix.
Conclusion: This is a rare case of appendiceal carcinoma invading the rectum and presenting as a positive screening fecal immunochemical test in an asymptomatic individual. We effectively demonstrate the use of preoperative MRI to identify the appendiceal origin of the tumor, as well as to demonstrate the extent of tumor spread, which assisted with operative management and treatment planning.
{"title":"Bladder-Adherent Primary Appendiceal Carcinoma Masquerading as a Carpeting Rectal Lesion Detected by a Fecal Immunochemical Test: A Case Report.","authors":"Nishigandha Burute, Olexiy Aseyev, Fauzia Hasan, Erika Allard-Ihala, Walid Shahrour, Ahmed Kotb, William Harris, Radu Rozenberg","doi":"10.1159/000535273","DOIUrl":"10.1159/000535273","url":null,"abstract":"<p><strong>Introduction: </strong>Primary appendiceal carcinoma is rare and comprises up to 1% of all colorectal malignancies. Its invasion into adjacent organs, such as the bladder and rectum, especially as a presenting characteristic, is even less common.</p><p><strong>Case presentation: </strong>A 75-year-old asymptomatic male tested positive on a screening fecal immunochemical test (FIT). Colonoscopy showed a rectosigmoid tumor and normal appendiceal orifice. Staging MRI surprisingly showed that the cancer was, in fact, of appendiceal origin, coursed posteriorly to invade the rectosigmoid and form adhesions with the urinary bladder. Staging CT did not show metastatic disease. Low anterior resection, en bloc appendectomy, and right hemicolectomy were performed along with cystectomy and ileal conduit. Hematoxylin and eosin stains showed appendiceal adenocarcinoma invading through the appendiceal wall into the rectal muscularis and submucosa. Features of neuroendocrine carcinoma were not identified on immunohistochemistry. This was a colonic type of adenocarcinoma of the appendix.</p><p><strong>Conclusion: </strong>This is a rare case of appendiceal carcinoma invading the rectum and presenting as a positive screening fecal immunochemical test in an asymptomatic individual. We effectively demonstrate the use of preoperative MRI to identify the appendiceal origin of the tumor, as well as to demonstrate the extent of tumor spread, which assisted with operative management and treatment planning.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-01-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10766397/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139377234","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 : 2023-12-18eCollection Date: 2023-01-01DOI: 10.1159/000535477
Brian Nohomovich, Minh H N Nguyen, Joseph Fakhoury, Russell C Cameron, Theotonius Gomes
Introduction: Down syndrome is due to trisomy 21 and is characterized by intellectual disability, dysmorphic facial features, congenital malformations, and gastrointestinal abnormalities. There is an increased appreciation of congenital portosystemic shunts in Down syndrome patients. Congenital portosystemic shunts have been associated with many defects in body systems, including cardiac, metabolic, and neurological.
Case presentation: Herein, we describe a portosystemic shunt in a Down syndrome patient that resulted in hyperammonemia with altered mental status and choreiform movements. Computed tomography angiography of the abdomen and pelvis identified a connection between the right portal vein and inferior vena cava. An 18 mm Amplatzer PFO closure device was placed within the congenital shunt, significantly improving symptoms. The patient has no sequelae from the related shunt or the device at the 2-year follow-up. We extensively reviewed the literature and identified cases of portosystemic shunts in Down syndrome patients. Shunts can either be extrahepatic or intrahepatic and are classified by vasculature connections.
Conclusion: From our literature review and case presentation, we identify other conditions in patients, including cardiac and gastrointestinal defects. We then review the available treatment options, whether observation or surgical, depending on the patient's clinical picture.
{"title":"Down Syndrome Patients with Congenital Portosystemic Shunts: A Case Report and Review.","authors":"Brian Nohomovich, Minh H N Nguyen, Joseph Fakhoury, Russell C Cameron, Theotonius Gomes","doi":"10.1159/000535477","DOIUrl":"https://doi.org/10.1159/000535477","url":null,"abstract":"<p><strong>Introduction: </strong>Down syndrome is due to trisomy 21 and is characterized by intellectual disability, dysmorphic facial features, congenital malformations, and gastrointestinal abnormalities. There is an increased appreciation of congenital portosystemic shunts in Down syndrome patients. Congenital portosystemic shunts have been associated with many defects in body systems, including cardiac, metabolic, and neurological.</p><p><strong>Case presentation: </strong>Herein, we describe a portosystemic shunt in a Down syndrome patient that resulted in hyperammonemia with altered mental status and choreiform movements. Computed tomography angiography of the abdomen and pelvis identified a connection between the right portal vein and inferior vena cava. An 18 mm Amplatzer PFO closure device was placed within the congenital shunt, significantly improving symptoms. The patient has no sequelae from the related shunt or the device at the 2-year follow-up. We extensively reviewed the literature and identified cases of portosystemic shunts in Down syndrome patients. Shunts can either be extrahepatic or intrahepatic and are classified by vasculature connections.</p><p><strong>Conclusion: </strong>From our literature review and case presentation, we identify other conditions in patients, including cardiac and gastrointestinal defects. We then review the available treatment options, whether observation or surgical, depending on the patient's clinical picture.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2023-12-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10727516/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138798376","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}
Abstract Introduction Dapsone is known to cause drug-induced liver injury (DILI) but can rarely induce the formation of hepatic granulomas. We describe a patient with jaundice who demonstrated granulomas on liver biopsy in response to dapsone. Her symptoms were only evident once steroids, used to also treat her pyoderma gangrenosum, had been tapered. Case Presentation In this case, a 67-year-old female was hospitalized due to 1 day of jaundice. She had started dapsone and prednisone concurrently 7 weeks ago to treat her pyoderma gangrenosum. Steroids were discontinued 4 days prior to symptoms. Her laboratories were notable for newly elevated alkaline phosphatase (756 U/L), aspartate transaminase (199 U/L), alanine transaminase (273 U/L), and total bilirubin (12.6 mg/dL). Dapsone was held due to suspicion for DILI. A liver biopsy was performed and disclosed non-necrotizing hepatic granulomas. After infectious and autoimmune causes were excluded, dapsone was determined to be the cause of her hepatic granulomas. Her bilirubin and liver enzymes steadily normalized over the next 4 weeks following discontinuation of dapsone. Conclusion Thus, dapsone-related liver injury may present following a steroid taper if dapsone and steroids had been initially started together. Hepatic granulomas, though rare, can be seen when dapsone causes DILI.
{"title":"Dapsone-Induced Granulomatous Cholestatic Hepatitis Unmasked by Steroid Taper: A Case Report","authors":"Shahana Prakash, Matthew Gosse, Tomohiro Tanaka","doi":"10.1159/000534818","DOIUrl":"https://doi.org/10.1159/000534818","url":null,"abstract":"Abstract Introduction Dapsone is known to cause drug-induced liver injury (DILI) but can rarely induce the formation of hepatic granulomas. We describe a patient with jaundice who demonstrated granulomas on liver biopsy in response to dapsone. Her symptoms were only evident once steroids, used to also treat her pyoderma gangrenosum, had been tapered. Case Presentation In this case, a 67-year-old female was hospitalized due to 1 day of jaundice. She had started dapsone and prednisone concurrently 7 weeks ago to treat her pyoderma gangrenosum. Steroids were discontinued 4 days prior to symptoms. Her laboratories were notable for newly elevated alkaline phosphatase (756 U/L), aspartate transaminase (199 U/L), alanine transaminase (273 U/L), and total bilirubin (12.6 mg/dL). Dapsone was held due to suspicion for DILI. A liver biopsy was performed and disclosed non-necrotizing hepatic granulomas. After infectious and autoimmune causes were excluded, dapsone was determined to be the cause of her hepatic granulomas. Her bilirubin and liver enzymes steadily normalized over the next 4 weeks following discontinuation of dapsone. Conclusion Thus, dapsone-related liver injury may present following a steroid taper if dapsone and steroids had been initially started together. Hepatic granulomas, though rare, can be seen when dapsone causes DILI.","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2023-12-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138584406","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}