Anouk Lehmann, Juerg Vosbeck, Diego Kyburz, Petr Hruz, Jan Hendrik Niess
{"title":"Risankizumab and Certolizumab Pegol Dual-Targeted Therapy for Crohn's Disease and Axial Spondyloarthritis: A Case Report.","authors":"Anouk Lehmann, Juerg Vosbeck, Diego Kyburz, Petr Hruz, Jan Hendrik Niess","doi":"10.1159/000542759","DOIUrl":"10.1159/000542759","url":null,"abstract":"<p><strong>Introduction: </strong>Treatment of Crohn's disease (CD) and axial spondyloarthritis (axSpA) is challenging, with CD refractory to anti-TNF antibodies. Here, we present for the first time a case treated with dual-targeted therapy (DTT) using the anti-IL-23 monoclonal risankizumab and the anti-TNF antibody certolizumab pegol.</p><p><strong>Case presentation: </strong>Our patient initially presented with axSpA at the age of 27. Nine years later, CD was diagnosed by the age of 36. One year after the diagnosis of CD, a spontaneous ileal perforation occurred as part of a disease course refractory to multiple anti-TNF antibodies and intolerance to immunomodulators. However, the axSpA showed a response to the anti-TNF certolizumab pegol. After stopping certolizumab pegol, we enrolled the patient into the M15-991 induction trial (MOTIVATE) and the maintenance trial (FORTIFY) testing the anti-IL-23 antibody risankizumab versus placebo in CD with failure to prior biological therapy. As a result, risankizumab induced a CD response but failed to control the axSpA. Considering the CD refractory and the axSpA responding to anti-TNFs, we initiated a DTT with risankizumab and certolizumab pegol. Risankizumab and certolizumab pegol together improved both CD and axSpA. As adverse events, there were only two episodes of spontaneously resolving common colds during the 19-month reviewed period.</p><p><strong>Conclusion: </strong>DTT using risankizumab and certolizumab pegol is effective in CD and axSpA without serious adverse events in our patient. Combining biologicals that target specific pathways in immune-mediated diseases promises excellent potential in CD associated with extraintestinal manifestations.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"22-30"},"PeriodicalIF":0.5,"publicationDate":"2024-12-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11668535/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466976","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}
{"title":"Steroid-Refractory Immune-Related Hepatitis Caused by Pembrolizumab with Stage IVB Non-Small Cell Lung Cancer: A Case Report.","authors":"Tomomi Hamaguchi, Makoto Ueno, Satoshi Kobayashi, Shun Tezuka, Manabu Morimoto, Terufumi Kato, Haruhiro Saito, Shinya Sato, Junji Furuse, Shin Maeda","doi":"10.1159/000542598","DOIUrl":"10.1159/000542598","url":null,"abstract":"<p><strong>Introduction: </strong>We report the case of a man in his 50s with stage IVB non-small cell lung cancer who developed severe immune-related hepatitis caused by pembrolizumab.</p><p><strong>Case presentation: </strong>He received carboplatin, pemetrexed, and pembrolizumab as first-line therapy. After four courses, each of the triplet regimen and maintenance therapy with pemetrexed and pembrolizumab, the patient developed immune-related pneumonitis and colitis. Therefore, pemetrexed and pembrolizumab were discontinued, and 0.5 mg/kg/day prednisolone was started. Despite gradual reduction of the prednisolone to 15 mg/day along with resolution of the pneumonitis and colitis, hepatic dysfunction occurred (elevated serum bilirubin and transaminase levels). We made a diagnosis of immune-related hepatitis based on liver biopsy results and negative results for other causes, such as viral infection. We increased the prednisolone dose to 2 mg/kg/day; however, the hepatic dysfunction was not resolved. Upon sequential methylprednisolone pulse therapy (1,000 mg/day), mycophenolate mofetil, and azathioprine treatment, the hepatic dysfunction plateaued but was not resolved. The patient did not respond to steroids for immune-related hepatitis, developed infectious enteritis owing to a compromised state, and died of sepsis on day 107 after diagnosis of immune-related hepatitis.</p><p><strong>Conclusion: </strong>This case highlights the importance of early diagnosis of steroid-refractory disease, prompt initiation of immunosuppressive agents, and steroid dose reduction in such cases. The changes in liver function during steroid non-response and immunosuppressive drug induction in this case are valuable as a reference for future cases of immune-related adverse event hepatitis.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"14-21"},"PeriodicalIF":0.5,"publicationDate":"2024-12-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11666266/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466978","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}
David Huynh, Myat Myat Khaing, Richard Gareth Fernandes, Reuben Malloy, Lei Lin, Robert Gilmore, Nicole Walker, Emi Khoo, Jakob Begun
{"title":"Upadacitinib Was Administered as a Sequential Salvage Therapy for Acute Severe Ulcerative Colitis: A Case Report.","authors":"David Huynh, Myat Myat Khaing, Richard Gareth Fernandes, Reuben Malloy, Lei Lin, Robert Gilmore, Nicole Walker, Emi Khoo, Jakob Begun","doi":"10.1159/000542711","DOIUrl":"10.1159/000542711","url":null,"abstract":"<p><strong>Introduction: </strong>Acute severe ulcerative colitis (ASUC) represents a medical emergency associated with high mortality and morbidity. While corticosteroids are the primary treatment, cases that are unresponsive often require rescue therapy with either infliximab or cyclosporine to reduce the rate of colectomy. Janus kinase inhibitors, such as tofacitinib and upadacitinib, are a highly efficacious therapy with rapid induction of clinical response in moderate to severe ulcerative colitis (UC). Limited data are available on its use on ASUC. We present the first case utilizing upadacitinib as sequential medical rescue therapy in ASUC as well as intestinal ultrasound as a useful tool for disease and response monitoring.</p><p><strong>Case presentation: </strong>A 69-year-old female who presented with corticosteroid-refractory ASUC partially responded to dose-intensified infliximab and finally achieved clinical remission with upadacitinib. This resulted in swift clinical remission and significant improvement in her mucosal inflammation on intestinal ultrasound.</p><p><strong>Conclusion: </strong>This successful intervention not only avoided colectomy but demonstrated sustained clinical and sonographic remission 16 weeks of post-treatment. Upadacitinib, with its rapid action and efficacy, shows promise in ASUC and should be supported by registration trials and real-world studies. Despite successful outcomes in this case, further validation and long-term data are necessary.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"1-6"},"PeriodicalIF":0.5,"publicationDate":"2024-12-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11666260/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466980","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}
Jelmer E Oor, Eline Groeneweg, Gijs C Bloemsma, Reinoud P H Bokkers, Joost M Klaase
{"title":"Endovascular Management of a Portal Vein Pseudoaneurysm following Pancreatoduodenectomy: A Case Report and Review of Literature.","authors":"Jelmer E Oor, Eline Groeneweg, Gijs C Bloemsma, Reinoud P H Bokkers, Joost M Klaase","doi":"10.1159/000542585","DOIUrl":"10.1159/000542585","url":null,"abstract":"<p><strong>Introduction: </strong>Portal vein pseudoaneurysm is a rare but potential life-threatening complication following pancreatoduodenectomy. We herein report on the successful, minimally invasive treatment of this serious complication.</p><p><strong>Case presentation: </strong>A 68-year-old male patient who had undergone pancreatoduodenectomy with portal vein wedge resection at another facility presented to our department due to persistent bile leakage and intermittent bleeding. Abdominal computed tomography scanning demonstrated a large fluid collection surrounding the pancreatojejunostomy, with an occlusion of the common hepatic artery as well as a portal vein pseudoaneurysm. Treatment was performed by means of a minimally invasive approach, including endovascular portal vein stent placement and percutaneous transhepatic biliary drainage (PTBD) through the dehiscent hepaticojejunostomy. This was followed by the placement of a percutaneous pigtail in the peripancreatic fluid collection for adequate drainage. Antibiotics were administered for a total duration of 6 weeks following stent placement. In the subsequent weeks, the PTBD could be internalized and the pigtail removed, after which patient was discharged. During outpatient visits, a contrast injection through the PTBD confirmed the absence of bile leakage, after which the drain could be removed.</p><p><strong>Conclusion: </strong>This article presents one of the few published cases of portal vein pseudoaneurysm following pancreatoduodenectomy and underscores the vital role of minimally invasive endovascular stent and PTBD placement in managing this rare and potentially lethal complication.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"7-13"},"PeriodicalIF":0.5,"publicationDate":"2024-12-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11666264/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467044","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}
{"title":"Gastrointestinal Neuroectodermal Tumor/Extraskeletal Ewing Sarcoma of the Ileum with Ulcerative Colitis.","authors":"Tianyu She, Stephanie Ren, Seymour Katz","doi":"10.1159/000542659","DOIUrl":"10.1159/000542659","url":null,"abstract":"<p><strong>Introduction: </strong>Malignant gastrointestinal neuroectodermal tumor (GNET), previously known as clear cell sarcoma-like tumor of the gastrointestinal tract, is an extremely rare and aggressive mesenchymal neoplasm characterized by high rates of recurrence, metastases, and mortality. Currently, there are no standardized guidelines for therapy.</p><p><strong>Case presentation: </strong>We present a case of GNET in a 32-year-old male with a history of lymphoma and ulcerative colitis (UC), who also had synchronous multiple liver metastasis. To our knowledge, this is the first documented case of GNET in a patient with inflammatory bowel disease.</p><p><strong>Conclusion: </strong>The narrow time frame in which UC and GNET were diagnosed warrants further investigation into their potential relationship.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"449-453"},"PeriodicalIF":0.5,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11637492/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466948","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}
{"title":"Acute Gastric Dilatation: A Retrospective Case Series from a Single Institution.","authors":"Lefika Bathobakae, Rammy Bashir, Sophia Venero, Tyler Wilkinson, Ruhin Yuridullah, Yana Cavanagh, Walid Baddoura","doi":"10.1159/000541516","DOIUrl":"https://doi.org/10.1159/000541516","url":null,"abstract":"<p><strong>Introduction: </strong>Acute gastric dilatation (AGD) is a massive distension of the stomach caused by the accumulation of gas, gastric secretions, or food material. AGD is a radiological diagnosis with no clear etiopathogenesis and is often misdiagnosed owing to a lack of clear diagnostic criteria and physician awareness.</p><p><strong>Case presentation: </strong>In this case series, we describe the clinical presentations and outcomes of 4 patients with AGD. Three (75%) of the patients were female, and one (25%) was male. The patients' ages ranged from 53 to 84 years, with an average age of 73.5 years. Abdominal pain, nausea, and vomiting were the most frequently reported complaints. Two (50%) patients had cancer, one (25%) had an acquired duodenal stenosis, and the fourth patient experienced an ileus.</p><p><strong>Conclusion: </strong>AGD is a surgical emergency with a 50-100% mortality rate; thus, prompt diagnosis and management are crucial. Herein, we describe a case series of AGDs that were diagnosed and managed at our institution. We aim to raise awareness about this fatal yet underrecognized clinical entity.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"439-448"},"PeriodicalIF":0.5,"publicationDate":"2024-10-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521523/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142543901","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}
{"title":"Colonic Fishbone-Induced Perforation Involving a Penile Colorectal Carcinoma: A Case Report.","authors":"Akira Imoto, Yuki Nagata, Yoshinori Shinohara, Hirota Miyazaki, Masanobu Fukumoto, Hidehiro Kitae, Masateru Asada, Masafumi Noda, Kaoru Okada, Akira Asai, Kazuhide Higuchi, Hiroki Nishikawa","doi":"10.1159/000541081","DOIUrl":"https://doi.org/10.1159/000541081","url":null,"abstract":"<p><strong>Introduction: </strong>Fishbone (FB) ingestion is a rare cause of gastrointestinal perforation. Herein, we report a case of FB-induced colonic perforation, in which the presence of a penile colonic carcinoma may have contributed to the development of the perforation.</p><p><strong>Case presentation: </strong>An 83-year-old man was admitted to our hospital with severe abdominal pain during bowel movement. Computed tomography (CT) yielded a diagnosis of sigmoid colonic perforation due to FB and secondary peritonitis. Preoperative endoscopic examination suggested that the perforation was associated with a stalked colon tumor in the vicinity. After undergoing low anterior resection and sigmoid colostomy, the patient is currently doing well.</p><p><strong>Conclusion: </strong>The incidence of FB-induced colorectal-cancer-related perforation is expected to increase in the future owing to an aging society, the increase in the rates of colorectal cancer, and increase in fish consumption. This rare case suggests that preoperative examinations are important and that even relatively small polyps can contribute to gastrointestinal perforation caused by FBs. Older individuals should exercise caution during fish ingestion.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"431-438"},"PeriodicalIF":0.5,"publicationDate":"2024-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521539/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142543902","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}
Daniella Soussi, Batool Helmi Ahmad Alharahsheh, Piers Robert Boshier, Jonathan Hoare, Natalie Direkze, Robert Thomas, Christopher John Peters, Sophie Stevens
{"title":"Endoluminal Vacuum Therapy for the Management of Boerhaave Syndrome: A Case Series.","authors":"Daniella Soussi, Batool Helmi Ahmad Alharahsheh, Piers Robert Boshier, Jonathan Hoare, Natalie Direkze, Robert Thomas, Christopher John Peters, Sophie Stevens","doi":"10.1159/000540694","DOIUrl":"https://doi.org/10.1159/000540694","url":null,"abstract":"<p><strong>Introduction: </strong>Boerhaave syndrome is a rare condition associated with high morbidity and mortality. Prompt intervention greatly improves outcomes, with surgery traditionally being the mainstay of management. Recent advances in therapeutic endoscopy have led to increasing interest in endoluminal vacuum therapy (EVT), a minimally invasive technique, allowing wound debridement and drainage, encouraging granulation tissue formation. EVT has been associated with positive clinical outcomes, including lower mortality rates compared to surgery and stenting for the management of anastomotic leaks, and to a lesser extent, oesophageal perforations. EVT has been adopted into practice across Europe; however, only few cases have been reported from the UK.</p><p><strong>Case presentations: </strong>We report three cases of Boerhaave syndrome, successfully managed with EVT, using the Eso-SPONGE <sup>®</sup> (B.Braun Medical Ltd, Sheffield, UK). EVT involves the placement of a polyurethane sponge into the wound cavity. The cavity is initially assessed, then an overtube is introduced through which the sponge is inserted, and then the overtube is removed. Sponge position is confirmed and adjusted if necessary. The sponge is connected via a trans-nasal drain to continuous negative pressure suction and is changed every 3-5 days. Having been deemed surgically unfit, all 3 patients were referred for EVT. All patients made excellent recovery and were discharged home.</p><p><strong>Conclusion: </strong>EVT is an effective management strategy for surgically unfit Boerhaave syndrome patients. Eso-SPONGE use aided drainage of the septic focus and closure of the defect, leading to complete recovery. Our findings support the existing evidence that EVT is a promising solution for Boerhaave syndrome.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"402-411"},"PeriodicalIF":0.5,"publicationDate":"2024-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521460/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142543903","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}
Billy Zhao, Hyun Jae Kim, Jessica Farrell, Wei Xiong, Jennifer Telford, Sarvee Moosavi
{"title":"Esophageal Tuberculosis as a Rare Cause of Dysphagia: Case Report.","authors":"Billy Zhao, Hyun Jae Kim, Jessica Farrell, Wei Xiong, Jennifer Telford, Sarvee Moosavi","doi":"10.1159/000540292","DOIUrl":"10.1159/000540292","url":null,"abstract":"<p><strong>Introduction: </strong>The esophagus and duodenum are rare sites of manifestation for extrapulmonary tuberculosis (TB). Its rarity makes the diagnosis challenging, especially when no other organ is involved, and the endoscopic findings may resemble malignancy.</p><p><strong>Case presentation: </strong>We report a unique case of a 37-year-old woman who presented with dysphagia secondary to esophageal TB with an endoscopic appearance of a submucosal mass resembling malignancy.</p><p><strong>Conclusion: </strong>Esophageal TB is a rare cause of dysphagia, especially in a western setting. It should always be considered as a potential etiology in patients with dysphagia.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"18 1","pages":"395-401"},"PeriodicalIF":0.5,"publicationDate":"2024-08-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324283/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981731","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}