Case Reports in Gastroenterology最新文献

{"title":"Acute Liver Failure due to Wilson's Disease and Rescue Therapy with Plasma Exchange: A Case Report and Literature Review.","authors":"Sameera Hajijama, Mahmoud Marashi, Samuel B Ho","doi":"10.1159/000544927","DOIUrl":"10.1159/000544927","url":null,"abstract":"<p><strong>Introduction: </strong>Wilson's disease (WD) is a rare genetic condition characterized by impaired copper metabolism and can rarely present as acute liver failure with an associated high mortality rate. Treatment often requires liver transplantation, and few rescue treatments have been shown to be effective by randomized trials. This is a case report and scoping literature review on plasmapheresis in acute liver failure due to WD.</p><p><strong>Case presentation: </strong>We report a case of a previously undiagnosed 32-year-old female who presented with acute liver failure due to WD. Initial severity indices (MELD = 31 and RWPI - Revised Wilson Prognostic Index = 14) warranted liver transplant referral; however, no facilities were available. The patient was managed by two sessions of plasmapheresis with plasma exchange with persistent improvement in MELD score over 1 week. Subsequent ongoing chelation therapy resulted in a total reversal of clinical liver disease over 2 years. Literature review resulted in 74 patients reported to have been treated with plasmapheresis for WD and acute liver failure. Of these, 40% survived without transplant. Out of the total patients reported, 54 patients had an available NWI score, with 46 patients having a high-risk score of ≥11 (85%) and 8 patients had a score <11 (15%). Of the 46 patients with a score ≥11, 21 (46%) recovered without liver transplantation, 16 (35%) underwent liver transplantation, 8 (17%) died of liver failure, and 1 (∼2%) died of sepsis. Of the 8 patients with a score of <11, 4 (50%) recovered without a liver transplantation, and 4 (50%) underwent transplantation.</p><p><strong>Conclusion: </strong>The outcomes observed in the literature and the favorable outcome of our patient suggest an emphasis on the use of urgent plasmapheresis with plasma exchange as an initial intervention for acute liver failure in WD, although further randomized controlled trials are needed for determining the optimal dose and duration of treatment.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"340-351"},"PeriodicalIF":0.5,"publicationDate":"2025-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12074618/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143977721","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":"Ischemic Colitis and Small Bowel Ischemia in a Vaccinated Patient with Mild COVID-19 Infection: A Case Report.","authors":"Maryam Aleissa, Ernesto Drelichman, Jasneet Bhullar","doi":"10.1159/000545495","DOIUrl":"https://doi.org/10.1159/000545495","url":null,"abstract":"<p><strong>Introduction: </strong>COVID-19 infection can cause bowel ischemia, with an incidence ranging from 0.22% to 10.5. The COVID-19 vaccine reduces respiratory symptoms and the need for cardiopulmonary support. However, its effects on other manifestations, such as bowel ischemia, have not been extensively studied. Despite having mild respiratory symptoms and receiving three doses of the COVID-19 vaccine, our patient developed ischemic colitis after her first infection and small bowel ischemia following her second infection.</p><p><strong>Case presentation: </strong>An 86-year-old woman presented to the emergency department (ED) with abdominal pain after a mild COVID-19 infection. She was admitted with ischemic colitis, but conservative treatment failed. In the operating room, her entire colon was found to be ischemic, necessitating a total colectomy with end ileostomy. Nine months later, she returned to the ED with mild respiratory symptoms and severe right upper quadrant pain. Imaging revealed pneumoperitoneum and a mid-abdominal abscess. An emergency laparotomy revealed small bowel perforation, with final histology confirming ischemia. The patient had received three doses of the COVID-19 vaccine and was on therapeutic anticoagulation for a history of pulmonary embolism.</p><p><strong>Conclusion: </strong>COVID-19 may increase the risk of bowel and colon ischemia even after vaccination. Patients presenting to the ED with severe abdominal pain and a recent COVID-19 infection should be carefully evaluated for ischemic events.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"335-339"},"PeriodicalIF":0.5,"publicationDate":"2025-05-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12064152/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143960854","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":"Pseudomelanosis Duodeni after Intravenous Iron Supplementation in a Patient with Multiple Comorbidities: A Case Report.","authors":"Olanrewaju Adeniran, Ayowumi Andrew Adekolu, Joshua Kirkpatrick, Ethan M Cohen, Abdullahi Adedotun Sulaiman, Farirai Marwizi, Zachary Kovac, Budoor Alqinai, Raja Samir Khan","doi":"10.1159/000545562","DOIUrl":"https://doi.org/10.1159/000545562","url":null,"abstract":"<p><strong>Introduction: </strong>Pseudomelanosis duodeni (PD) is a rare condition characterized by multiple pigmented speckles in the duodenum. It is typically seen in adults and has been associated with chronic kidney disease, hypertension, iron deficiency anemia, diabetes mellitus, and the use of medications like oral iron, hydralazine, furosemide, and hydrochlorothiazide. While oral iron is common, only one documented PD has been associated with intravenous (IV) iron supplementation.</p><p><strong>Case presentation: </strong>A 69-year-old female with multiple comorbidities, including stage 4 chronic kidney disease and hypertension, presented with persistent nausea and vomiting. An esophagogastroduodenoscopy (EGD) revealed diffuse, dark speckling throughout the duodenal mucosa. Biopsies confirmed PD through Perl Prussian blue and Fontana-Masson staining, which detected iron in black/brown pigmentations within macrophages. The patient received a 5-week course of weekly intravenous iron supplementation 6 months before presentation. Previous EGD 7 and 5 years earlier showed no evidence of PD despite the patient being on sulfur-containing antihypertensive medications. This case suggests a likely interplay between IV iron and sulfur-containing medications in PD development.</p><p><strong>Conclusion: </strong>While PD has been associated with multiple comorbidities and certain medications, the causal mechanism remains unclear. PD likely entails defective iron transport and iron sulfide accumulation within duodenal macrophages. This case highlights the potential role of IV iron supplementation in PD development, even in the absence of oral iron intake. PD is regarded as a benign condition with no specific guidelines for treatment or follow-up, but a biopsy is crucial to rule out other differential diagnoses and avoid unwanted interventions.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"328-334"},"PeriodicalIF":0.5,"publicationDate":"2025-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12058112/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143981781","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":"Vancomycin Therapy for Induction and Maintenance of Remission in a Patient with Refractory Ulcerative Colitis and Primary Sclerosing Cholangitis: A Case Report and Literature Review.","authors":"Naama Lang, Yana Davidov, Tomer Hoffman, Sivan Harnik, Abraham Rami Eliakim, Shomron Ben-Horin, Bella Ungar","doi":"10.1159/000544161","DOIUrl":"https://doi.org/10.1159/000544161","url":null,"abstract":"<p><strong>Introduction: </strong>Ulcerative colitis is a chronic inflammatory bowel disease that affects the colon. About 5% of ulcerative colitis patients also present with primary sclerosing cholangitis, a chronic inflammatory disease marked by cholestasis and progressive fibrosis of the bile ducts, and results in the necessity of liver transplantation. Ulcerative colitis treatment in primary sclerosing cholangitis patients is challenging due to potential resistance to conventional therapies.</p><p><strong>Case presentations: </strong>We describe a patient with ulcerative colitis and primary sclerosing cholangitis, whose ulcerative colitis relapsed following a liver transplant. After failure of conventional treatment and further deterioration in her colitis, with negative clostridium difficile, she was treated with vancomycin with beneficial long-term clinical and endoscopic responses.</p><p><strong>Conclusion: </strong>This case report, along with others we reviewed, suggests that vancomycin treatment should be considered as a treatment for ulcerative colitis and primary sclerosing cholangitis patients after conventional therapies for the colitis prove ineffective.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"320-327"},"PeriodicalIF":0.5,"publicationDate":"2025-05-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12054989/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143978365","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":"Cap Polyposis Syndrome Imitating Inflammatory Bowel Disease with Polyps Extending to the Terminal Ileum.","authors":"Garrett Thomas Coleman, Rawan Dayah, Suimin Qiu, Gurinder Luthra","doi":"10.1159/000545182","DOIUrl":"https://doi.org/10.1159/000545182","url":null,"abstract":"<p><strong>Introduction: </strong>Cap polyposis syndrome is a rare subtype of mucosal prolapse disease characterized by erythematous, inflammatory colonic polyps covered by a cap of fibrinopurulent mucous. Although a benign condition, patients may present with significant symptoms that can be suggestive of inflammatory bowel disease or colorectal cancer.</p><p><strong>Case presentation: </strong>We describe the case of a 34-year-old male who presented with a 5-month history of diarrhea and 40-lb weight loss following hospitalization for enterotoxigenic <i>Escherichia coli</i> colitis. The patient had a past medical history significant for prior colonoscopy revealing hundreds of polyps and a father who died of colorectal cancer at age 45. Multiple repeat infectious stool workups were negative, and antibiotics failed to resolve the patient's symptoms. The patient underwent endoscopy which revealed numerous polyps from the rectum to the terminal ileum that appeared similarly to pseudopolyps giving concern for inflammatory bowel disease. Subsequent histology demonstrated surface erosion and inflammation without dysplasia. Review of endoscopy showed inflammatory polyps with a cap of fibrinopurulent mucous. In the absence of chronic inflammation (C-reactive protein was within normal limits following hospitalization), endoscopic and histologic findings were suggestive of cap polyposis syndrome.</p><p><strong>Conclusion: </strong>Cap polyposis is diagnosed endoscopically and histologically. While most cases of cap polyposis are confined to the distal colon and rectum, we believe that this is the first case of cap polyposis syndrome extending to the terminal ileum. Treatment of cap polyposis syndrome is dependent on the severity of symptoms.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"314-319"},"PeriodicalIF":0.5,"publicationDate":"2025-05-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12052363/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143979024","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":"Erratum.","authors":"","doi":"10.1159/000545486","DOIUrl":"https://doi.org/10.1159/000545486","url":null,"abstract":"<p><p>[This corrects the article DOI: 10.1159/000544099.].</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"313"},"PeriodicalIF":0.5,"publicationDate":"2025-04-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12043278/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143986138","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":"Autoimmune Pancreatitis in Children: A Case Report.","authors":"Barbara Hendlisz, Amélie Dirckx, Marie Cassart, Phu Quoc Lê","doi":"10.1159/000544785","DOIUrl":"https://doi.org/10.1159/000544785","url":null,"abstract":"<p><strong>Introduction: </strong>Autoimmune pancreatitis (AIP) is a rare and distinct condition in children compared to adults. Despite being based on adult diagnostic criteria, several differences have been reported, making pediatric AIP a challenging diagnosis.</p><p><strong>Case presentation: </strong>A 14-year-old adolescent presented to the emergency department with abdominal pain, vomiting, and jaundice. A combination of biological and radiological assessments led to the diagnosis of AIP. The patient experienced a swift and complete recovery without intensive treatment.</p><p><strong>Conclusion: </strong>AIP in children is an intriguing diagnosis and should be considered when a child presents with abdominal pain, vomiting, and jaundice. The natural history and pathophysiology of the disease are still uncertain. Specific markers for the disease need to be established. Studies on larger sample sizes are necessary to address these questions and improve AIP diagnosis and management.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"303-312"},"PeriodicalIF":0.5,"publicationDate":"2025-04-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12040306/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143960192","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":"An Unusual Complication of Diverticular Bleed: Dysphagia and Food Impaction from Black Esophagus.","authors":"Nicholas Noverati, Beatriz Torre, Maria Mostyka, Cuckoo Choudhary, Christopher Henry, David Kastenberg, Stephanie Moleski","doi":"10.1159/000545171","DOIUrl":"https://doi.org/10.1159/000545171","url":null,"abstract":"<p><strong>Introduction: </strong>Acute esophageal necrosis, or black esophagus, is a rare clinical phenomenon typically seen in older men after an acute triggering event. It may present with dysphagia due to stricturing or dysmotility, a complication of severe esophageal inflammation.</p><p><strong>Case presentation: </strong>Here we describe a case of a woman with several cardiovascular risk factors who developed black esophagus following diverticular-related hemorrhagic shock and presented with chest pain followed by dysphagia and food impaction likely as a result of mucosal sloughing and altered esophageal motility. The diagnosis was confirmed endoscopically and pathology revealed coagulative necrosis due to ischemia.</p><p><strong>Discussion: </strong>Acute esophageal necrosis should be suspected in patients with cardiovascular risk factors and hemodynamic compromise and may be confirmed with endoscopy. As dysphagia may complicate this condition, slow advancement of diet while healing is advised.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"298-302"},"PeriodicalIF":0.5,"publicationDate":"2025-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12037161/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143966440","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":"Filgotinib to Treat Acute Severe Refractory Ulcerative Colitis: A Case Report and Review of the Literature.","authors":"Giuliana Vespere, Silvia Sedda, Pasquale Madonna, Roberta Abete, Alfredo D'Avino, Manuela Maione, Marina Lugarà, Antonietta Tazza, Franco Scaldaferri, Giuseppe Vitiello, Leonardo De Luca","doi":"10.1159/000545263","DOIUrl":"https://doi.org/10.1159/000545263","url":null,"abstract":"<p><strong>Introduction: </strong>Acute severe ulcerative colitis (ASUC) is a serious complication affecting 9%-15% of patients with UC within 3 months of diagnosis and up to 28% of patients during the course of the disease. Despite the use of infliximab and calcineurin inhibitors, the rate of colectomy remains high both during the hospitalization and in the 5 years after an acute episode. We present a case of ASUC that was unresponsive to conventional therapies but was successfully treated with filgotinib.</p><p><strong>Case presentation: </strong>A 21-year-old male with a recent diagnosis of UC presented to our hospital with a severe flare. He received rescue therapy of high-dose intravenous steroids and 10 mg/kg infliximab. We observed little clinical and biochemical benefits. The patient declined the surgical option. Therefore, we decided to start a second rescue therapy with a new and rapid-acting Janus kinase inhibitor, filgotinib, due to its characteristics and pharmacokinetic profile of rapid absorption and metabolism. The patient showed an immediate clinical and biochemical response at 48 h, an endoscopic response at week 3, and an endoscopic remission at week 10. No recurrence was observed after 12 months of follow-up. The patient is in clinical remission with a good quality of life.</p><p><strong>Conclusion: </strong>Filgotinib may be an effective second-line therapy in an emergency setting such as ASUC in patients unresponsive to conventional therapy.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"289-297"},"PeriodicalIF":0.5,"publicationDate":"2025-04-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12028979/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143986140","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":"Jejunal Lipoma-Induced Intussusception Mimicking Crohn's Disease: A Case Report.","authors":"Naveena Luke, Tianyu She, Divya Roy, Toyooki Sonoda, Seymour Katz","doi":"10.1159/000545297","DOIUrl":"https://doi.org/10.1159/000545297","url":null,"abstract":"<p><strong>Introduction: </strong>Chronic gastrointestinal bleeding in patients with Crohn's disease presents diagnostic challenges. Adult intussusception is rare and typically caused by a pathological lead point, such as a tumor or inflammatory lesion. Lipomas, though benign, can lead to obstruction and bleeding, requiring differentiation from inflammatory causes for appropriate management.</p><p><strong>Case presentation: </strong>A 70-year-old male with Crohn's disease and chronic anemia presented with recurrent obscure gastrointestinal bleeding. Initial endoscopy was unremarkable, but capsule endoscopy identified a bleeding jejunal lesion. Double-balloon enteroscopy and imaging confirmed a jejunal lipoma causing intermittent intussusception. Due to persistent anemia, the patient underwent laparoscopic resection, with pathology confirming an ulcerated lipoma. His anemia resolved postoperatively.</p><p><strong>Conclusion: </strong>This case underscores the importance of considering structural lesions like lipomas in patients with chronic bleeding and Crohn's disease. A multimodal approach, including advanced imaging and enteroscopy, is crucial for accurate diagnosis and management. Surgical resection remains the preferred treatment for symptomatic small bowel lipomas.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"282-288"},"PeriodicalIF":0.5,"publicationDate":"2025-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12017744/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143965167","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}