Introduction: High-grade appendiceal mucinous neoplasms (HAMNs) are rare mucin-producing epithelial tumors of the appendix, typically seen in middle-aged to elderly individuals. They are defined by high-grade cytologic atypia and complex architecture but without infiltrative invasion. Occurrence in pediatric or adolescent patients is virtually unknown.
Case presentation: We report a 16-year-old female who presented with signs and symptoms suggestive of acute appendicitis. Ultrasonography supported the diagnosis. A laparoscopic appendectomy was performed. Gross examination revealed a dilated appendix with mucoid contents. Histopathological evaluation confirmed HAMN confined to the appendix.
Conclusion: To our knowledge, this is the second reported case of HAMN in an adolescent. This case underscores the importance of histopathologic evaluation of all appendectomy specimens, even in young patients, due to the possibility of underlying neoplasia.
{"title":"A Diagnostic Dilemma: High-Grade Appendiceal Mucinous Neoplasm Presenting as Acute Appendicitis in a 16-Year-Old Female - Case Report and Literature Review.","authors":"Madhuri Singh, Charusheela Gore, Sushama Gurwale, Jhasnavi Mandala, Apoorva Makan","doi":"10.1159/000550074","DOIUrl":"https://doi.org/10.1159/000550074","url":null,"abstract":"<p><strong>Introduction: </strong>High-grade appendiceal mucinous neoplasms (HAMNs) are rare mucin-producing epithelial tumors of the appendix, typically seen in middle-aged to elderly individuals. They are defined by high-grade cytologic atypia and complex architecture but without infiltrative invasion. Occurrence in pediatric or adolescent patients is virtually unknown.</p><p><strong>Case presentation: </strong>We report a 16-year-old female who presented with signs and symptoms suggestive of acute appendicitis. Ultrasonography supported the diagnosis. A laparoscopic appendectomy was performed. Gross examination revealed a dilated appendix with mucoid contents. Histopathological evaluation confirmed HAMN confined to the appendix.</p><p><strong>Conclusion: </strong>To our knowledge, this is the second reported case of HAMN in an adolescent. This case underscores the importance of histopathologic evaluation of all appendectomy specimens, even in young patients, due to the possibility of underlying neoplasia.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"20 1","pages":"35-39"},"PeriodicalIF":0.6,"publicationDate":"2026-01-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12880843/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146140657","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-12-24eCollection Date: 2026-01-01DOI: 10.1159/000549935
Thaer Swaid, Carl Kassab, Najla Saleh Ben Ghashir, Saeed Husain Al Marzooqi
Introduction: Hepatic angiosarcoma (HA) is a rare endothelial malignancy with poor prognosis. Chronic Schistosoma infection causes periportal fibrosis and portal hypertension, but has not been established as a risk factor for hepatic vascular tumors. We report a case of high-grade HA coexisting with chronic schistosomiasis in a patient presenting with acute decompensated cirrhosis.
Case presentation: A 64-year-old Filipino woman with no known chemical exposures presented with progressive abdominal pain, jaundice, ascites, and hepatic encephalopathy. Doppler ultrasound initially suggested Budd-Chiari syndrome, later excluded on advanced imaging. MRI showed multifocal hepatic lesions, and CT demonstrated multiple intra-abdominal hematomas without active bleeding. Liver biopsy confirmed poorly differentiated HA (CD31+/CD34+) and revealed calcified Schistosoma eggs. Her course was complicated by hemorrhagic shock after biopsy and paracentesis, spontaneous bacterial peritonitis, acute kidney injury, and worsening hepatic encephalopathy. She died despite aggressive supportive care.
Conclusion: This case illustrates the diagnostic challenges and rapid progression of HA, especially in coagulopathic patients where biopsy carries significant risk. The coexistence of chronic schistosomiasis raises the possibility of parasitic inflammation contributing to hepatic vascular tumorigenesis. HA should be considered among unexplained hepatic masses with atypical imaging and clinical features.
{"title":"Hepatic Angiosarcoma Associated with Chronic Schistosomiasis in a Patient with Decompensated Cirrhosis: A Rare Case Report.","authors":"Thaer Swaid, Carl Kassab, Najla Saleh Ben Ghashir, Saeed Husain Al Marzooqi","doi":"10.1159/000549935","DOIUrl":"10.1159/000549935","url":null,"abstract":"<p><strong>Introduction: </strong>Hepatic angiosarcoma (HA) is a rare endothelial malignancy with poor prognosis. Chronic Schistosoma infection causes periportal fibrosis and portal hypertension, but has not been established as a risk factor for hepatic vascular tumors. We report a case of high-grade HA coexisting with chronic schistosomiasis in a patient presenting with acute decompensated cirrhosis.</p><p><strong>Case presentation: </strong>A 64-year-old Filipino woman with no known chemical exposures presented with progressive abdominal pain, jaundice, ascites, and hepatic encephalopathy. Doppler ultrasound initially suggested Budd-Chiari syndrome, later excluded on advanced imaging. MRI showed multifocal hepatic lesions, and CT demonstrated multiple intra-abdominal hematomas without active bleeding. Liver biopsy confirmed poorly differentiated HA (CD31+/CD34+) and revealed calcified Schistosoma eggs. Her course was complicated by hemorrhagic shock after biopsy and paracentesis, spontaneous bacterial peritonitis, acute kidney injury, and worsening hepatic encephalopathy. She died despite aggressive supportive care.</p><p><strong>Conclusion: </strong>This case illustrates the diagnostic challenges and rapid progression of HA, especially in coagulopathic patients where biopsy carries significant risk. The coexistence of chronic schistosomiasis raises the possibility of parasitic inflammation contributing to hepatic vascular tumorigenesis. HA should be considered among unexplained hepatic masses with atypical imaging and clinical features.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"20 1","pages":"29-34"},"PeriodicalIF":0.6,"publicationDate":"2025-12-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12830003/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146050262","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: The incidence of disseminated carcinomatosis of the bone marrow (DCBM) associated with colorectal cancer is low, but it can rapidly progress and cause even death. A previous study has suggested a relationship between mucin profiles and the prognosis of colorectal cancer. However, there are no reports of mucin staining in cases of DCBM with post-colonoscopy colorectal cancer (PCCRC).
Case presentation: A 58-year-old man who had undergone colonoscopy (CS) 1 year ago complained of fatigue and fever. The man was diagnosed with virus-associated hemophagocytic syndrome and received steroid pulse therapy. Despite treatment, his condition deteriorated. 18F-fluorodeoxyglucose positron emission tomography-computed tomography revealed diffuse uptake in the bone marrow. A bone marrow biopsy revealed atypical CK20-positive and CK7-negative cellular nests, suggesting a primary gastrointestinal tumor. Subsequent CS at our hospital revealed a 30-mm type 4 transverse colorectal cancer lesion. Thus, the patient was diagnosed with DCBM associated with PCCRC. The patient died 18 days after his last CS. Mucin staining revealed that the cancer cells were MUC2-positive and MUC5AC-negative. MUC5AC negativity has been associated with poor prognosis in colorectal cancer and may have suggested rapid disease progression.
Conclusion: The mucin staining result of MUC5AC negativity suggested rapid disease progression and poor prognosis in this case.
{"title":"Immunohistochemical Staining for Disseminated Carcinomatosis of the Bone Marrow in a Patient with Post-Colonoscopy Colorectal Cancer: A Case Report.","authors":"Tomoaki Mochimaru, Kazumasa Kawashima, Michio Onizawa, Yu Watahiki, Yosuke Takahata, Mai Murakami, Kakeru Otomo, Tomoyuki Asano, Shuhei Yoshida, Satoshi Kawana, Naohiko Gunji, Yuko Hashimoto, Takuto Hikichi, Hiromasa Ohira","doi":"10.1159/000549280","DOIUrl":"10.1159/000549280","url":null,"abstract":"<p><strong>Introduction: </strong>The incidence of disseminated carcinomatosis of the bone marrow (DCBM) associated with colorectal cancer is low, but it can rapidly progress and cause even death. A previous study has suggested a relationship between mucin profiles and the prognosis of colorectal cancer. However, there are no reports of mucin staining in cases of DCBM with post-colonoscopy colorectal cancer (PCCRC).</p><p><strong>Case presentation: </strong>A 58-year-old man who had undergone colonoscopy (CS) 1 year ago complained of fatigue and fever. The man was diagnosed with virus-associated hemophagocytic syndrome and received steroid pulse therapy. Despite treatment, his condition deteriorated. <sup>18</sup>F-fluorodeoxyglucose positron emission tomography-computed tomography revealed diffuse uptake in the bone marrow. A bone marrow biopsy revealed atypical CK20-positive and CK7-negative cellular nests, suggesting a primary gastrointestinal tumor. Subsequent CS at our hospital revealed a 30-mm type 4 transverse colorectal cancer lesion. Thus, the patient was diagnosed with DCBM associated with PCCRC. The patient died 18 days after his last CS. Mucin staining revealed that the cancer cells were MUC2-positive and MUC5AC-negative. MUC5AC negativity has been associated with poor prognosis in colorectal cancer and may have suggested rapid disease progression.</p><p><strong>Conclusion: </strong>The mucin staining result of MUC5AC negativity suggested rapid disease progression and poor prognosis in this case.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"777-783"},"PeriodicalIF":0.6,"publicationDate":"2025-12-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12705095/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145766996","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: Endoscopic ultrasound-guided pelvic abscess drainage (EUS-PAD) was reported for the first time in 2003. However, there were few reports focused on EUS-PAD in pediatric patients beforehand. This procedure is one of the minimally invasive methods of treating pelvic abscess, which is close to the rectum. Here, we report the cases of two pediatric patients who underwent EUS-PAD.
Case presentation: Two pediatric patients were referred to our hospital for the treatment of pelvic abscesses associated with appendicitis. Using endoscopic ultrasound (EUS) guidance, a pelvic abscess was identified adjacent to the rectal wall. After successive dilatation through a guidewire during fluoroscopic imaging, the deployment of one or two double pigtail stents was performed. Following successful stent deployment, both patients experienced good courses without any complications. After addressing the consequences of complicated appendicitis, both patients underwent laparoscopic appendectomy, which resulted in favorable postoperative courses.
Conclusions: EUS-guided drainage can be considered a safe and effective method for primarily dealing with pelvic abscesses and as an efficacious bridge-to-surgery strategy for pediatric patients.
{"title":"Endoscopic Ultrasound-Guided Pelvic Abscess Drainage Especially for Pediatric Patients: A Report of Two Cases.","authors":"Yusaku Yokotani, Hiroaki Kitae, Shun Takakura, Naoaki Akamatsu, Koichi Soga","doi":"10.1159/000550073","DOIUrl":"10.1159/000550073","url":null,"abstract":"<p><strong>Introduction: </strong>Endoscopic ultrasound-guided pelvic abscess drainage (EUS-PAD) was reported for the first time in 2003. However, there were few reports focused on EUS-PAD in pediatric patients beforehand. This procedure is one of the minimally invasive methods of treating pelvic abscess, which is close to the rectum. Here, we report the cases of two pediatric patients who underwent EUS-PAD.</p><p><strong>Case presentation: </strong>Two pediatric patients were referred to our hospital for the treatment of pelvic abscesses associated with appendicitis. Using endoscopic ultrasound (EUS) guidance, a pelvic abscess was identified adjacent to the rectal wall. After successive dilatation through a guidewire during fluoroscopic imaging, the deployment of one or two double pigtail stents was performed. Following successful stent deployment, both patients experienced good courses without any complications. After addressing the consequences of complicated appendicitis, both patients underwent laparoscopic appendectomy, which resulted in favorable postoperative courses.</p><p><strong>Conclusions: </strong>EUS-guided drainage can be considered a safe and effective method for primarily dealing with pelvic abscesses and as an efficacious bridge-to-surgery strategy for pediatric patients.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"20 1","pages":"23-28"},"PeriodicalIF":0.6,"publicationDate":"2025-12-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12823109/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146028403","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-12-12eCollection Date: 2025-01-01DOI: 10.1159/000549436
Terry R Wagner, Sem F Hardon, Frank C den Boer, Jorn P Meekel
Introduction: Foreign body ingestion is a common clinical occurrence, often resolving spontaneously as the object passes through the gastrointestinal tract. We present a unique case of sigmoid perforation with an associated local abscess, successfully managed with sigmoidectomy and direct anastomosis.
Case presentation: A 75-year-old male presented to the emergency department after accidentally ingesting his dentures. Conservative management was attempted initially. Four days later, the patient developed abdominal pain. Computed tomography scan revealed signs of perforation, and diagnostic laparoscopy confirmed a sigmoid perforation with a localized abscess. A sigmoidectomy with end-to-end circular stapled anastomosis was performed, during which the dentures were mobilized retrogradely to enable controlled extraction, leading to a full and rapid recovery.
Conclusion: This case highlights the potential complications and occasional need for surgery due to ingested foreign bodies and the importance of accurate diagnosis and vigilant monitoring in managing these patients.
{"title":"Denture Ingestion, Gastrointestinal Transit and Subsequent Complications in a 75-Year-Old Male: A Case Report.","authors":"Terry R Wagner, Sem F Hardon, Frank C den Boer, Jorn P Meekel","doi":"10.1159/000549436","DOIUrl":"10.1159/000549436","url":null,"abstract":"<p><strong>Introduction: </strong>Foreign body ingestion is a common clinical occurrence, often resolving spontaneously as the object passes through the gastrointestinal tract. We present a unique case of sigmoid perforation with an associated local abscess, successfully managed with sigmoidectomy and direct anastomosis.</p><p><strong>Case presentation: </strong>A 75-year-old male presented to the emergency department after accidentally ingesting his dentures. Conservative management was attempted initially. Four days later, the patient developed abdominal pain. Computed tomography scan revealed signs of perforation, and diagnostic laparoscopy confirmed a sigmoid perforation with a localized abscess. A sigmoidectomy with end-to-end circular stapled anastomosis was performed, during which the dentures were mobilized retrogradely to enable controlled extraction, leading to a full and rapid recovery.</p><p><strong>Conclusion: </strong>This case highlights the potential complications and occasional need for surgery due to ingested foreign bodies and the importance of accurate diagnosis and vigilant monitoring in managing these patients.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"767-772"},"PeriodicalIF":0.6,"publicationDate":"2025-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12700593/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145755429","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-12-04eCollection Date: 2026-01-01DOI: 10.1159/000548125
Christopher J Shephard, Xiaomin Ma, Rozemary Karamatic
Introduction: Percutaneous endoscopic gastrostomy (PEG) tubes are commonly used to facilitate long-term enteral nutrition. Selected PEG tubes may be appropriately removed without endoscopic guidance via gentle traction, which is seldom associated with major bleeding sequelae. Rare reports of major upper gastrointestinal (GI) hemorrhage following PEG tube removal have been managed using Foley catheter balloon tamponade or endoscopic over-the-scope clipping. Prompt reinsertion of a PEG tube may also be considered.
Case presentation: We present the case of a multi-morbid 73-year-old female who experienced life-threatening hemodynamically unstable upper GI bleeding following traction PEG tube removal, which proved refractory to standard endoscopic measures of adrenaline injection and hemostatic clipping, as well as rotational thromboelastometry-guided correction of consumptive coagulopathy. There was no suitable target for angioembolization. Ultimately, in the context of continued hemodynamically unstable GI bleeding, definitive surgical hemostasis - a mode of intervention not frequently reported for bleeding of this nature - was achieved during emergency laparotomy via complete excision of the PEG tract and wedge excision of the involved stomach wall.
Conclusion: Life-threatening hemodynamically unstable GI bleeding rarely complicates traction PEG tube removal. In elderly patients with multiple comorbidities, PEG tract bleeding refractory to standard endotherapy and not targetable by angioembolization can be safely and effectively managed with timely surgical intervention.
{"title":"Life-Threatening Upper Gastrointestinal Bleeding following Percutaneous Endoscopic Gastrostomy Tube Removal under Traction: A Case Report.","authors":"Christopher J Shephard, Xiaomin Ma, Rozemary Karamatic","doi":"10.1159/000548125","DOIUrl":"10.1159/000548125","url":null,"abstract":"<p><strong>Introduction: </strong>Percutaneous endoscopic gastrostomy (PEG) tubes are commonly used to facilitate long-term enteral nutrition. Selected PEG tubes may be appropriately removed without endoscopic guidance via gentle traction, which is seldom associated with major bleeding sequelae. Rare reports of major upper gastrointestinal (GI) hemorrhage following PEG tube removal have been managed using Foley catheter balloon tamponade or endoscopic over-the-scope clipping. Prompt reinsertion of a PEG tube may also be considered.</p><p><strong>Case presentation: </strong>We present the case of a multi-morbid 73-year-old female who experienced life-threatening hemodynamically unstable upper GI bleeding following traction PEG tube removal, which proved refractory to standard endoscopic measures of adrenaline injection and hemostatic clipping, as well as rotational thromboelastometry-guided correction of consumptive coagulopathy. There was no suitable target for angioembolization. Ultimately, in the context of continued hemodynamically unstable GI bleeding, definitive surgical hemostasis - a mode of intervention not frequently reported for bleeding of this nature - was achieved during emergency laparotomy via complete excision of the PEG tract and wedge excision of the involved stomach wall.</p><p><strong>Conclusion: </strong>Life-threatening hemodynamically unstable GI bleeding rarely complicates traction PEG tube removal. In elderly patients with multiple comorbidities, PEG tract bleeding refractory to standard endotherapy and not targetable by angioembolization can be safely and effectively managed with timely surgical intervention.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"20 1","pages":"16-22"},"PeriodicalIF":0.6,"publicationDate":"2025-12-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12810975/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145997379","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-11-24eCollection Date: 2025-01-01DOI: 10.1159/000548927
Chunxia Wang, Yu Zhong, Rihua Zheng
Introduction: Colonic mucosal pseudolipomatosis (CMP) is a rare mucosal lesion of the colon. Most patients are asymptomatic, while some present with abdominal pain, diarrhea, or bloating. However, cases complicated by hemorrhage are extremely rare and have not been previously reported. Endoscopically, CMP manifests as solitary or multiple scattered white plaques in intestinal segments, manifesting as a "snow-white sign."
Case presentation: This article reports a case of CMP with hemorrhage following colonoscopy.
Conclusion: With the increasing incidence of CMP in recent years, endoscopists have gained deeper insights into this lesion. The vigilance is warranted for its potential association with hemorrhage. In addition, in order to avoid such iatrogenic injury, endoscope disinfection monitoring should be paid more attention.
{"title":"Colonic Mucosal Pseudolipomatosis with Hemorrhage: A Case Report of Peracetic Acid-Induced Pathogenesis.","authors":"Chunxia Wang, Yu Zhong, Rihua Zheng","doi":"10.1159/000548927","DOIUrl":"10.1159/000548927","url":null,"abstract":"<p><strong>Introduction: </strong>Colonic mucosal pseudolipomatosis (CMP) is a rare mucosal lesion of the colon. Most patients are asymptomatic, while some present with abdominal pain, diarrhea, or bloating. However, cases complicated by hemorrhage are extremely rare and have not been previously reported. Endoscopically, CMP manifests as solitary or multiple scattered white plaques in intestinal segments, manifesting as a \"snow-white sign.\"</p><p><strong>Case presentation: </strong>This article reports a case of CMP with hemorrhage following colonoscopy.</p><p><strong>Conclusion: </strong>With the increasing incidence of CMP in recent years, endoscopists have gained deeper insights into this lesion. The vigilance is warranted for its potential association with hemorrhage. In addition, in order to avoid such iatrogenic injury, endoscope disinfection monitoring should be paid more attention.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"773-776"},"PeriodicalIF":0.6,"publicationDate":"2025-11-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12705116/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145766931","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-11-21eCollection Date: 2025-01-01DOI: 10.1159/000548860
Thaer Swaid, Layth Hamzeh, Maryam Al Ahmad, Sulaman Magdub, Ishtiaq Ahmed
Introduction: Eosinophilic esophagitis (EoE) is a chronic, immune-mediated esophageal disease characterized by eosinophilic infiltration of the esophageal mucosa, leading to symptoms of esophageal dysfunction. Common presentations include dysphagia, food impaction, and chest discomfort, often in patients with a history of atopy.
Case presentation: This case report highlights an atypical outcome of EoE, intramural esophageal dissection, in a young adult patient post-endoscopy.
Conclusion: This case report emphasizes the importance of early recognition and treatment to prevent long-term complications, both pathological and iatrogenic.
{"title":"Iatrogenic Intramural Esophageal Dissection in Eosinophilic Esophagitis: A Rare Complication of Diagnostic Endoscopy in a Young Atopic Patient - Case Report and Review.","authors":"Thaer Swaid, Layth Hamzeh, Maryam Al Ahmad, Sulaman Magdub, Ishtiaq Ahmed","doi":"10.1159/000548860","DOIUrl":"10.1159/000548860","url":null,"abstract":"<p><strong>Introduction: </strong>Eosinophilic esophagitis (EoE) is a chronic, immune-mediated esophageal disease characterized by eosinophilic infiltration of the esophageal mucosa, leading to symptoms of esophageal dysfunction. Common presentations include dysphagia, food impaction, and chest discomfort, often in patients with a history of atopy.</p><p><strong>Case presentation: </strong>This case report highlights an atypical outcome of EoE, intramural esophageal dissection, in a young adult patient post-endoscopy.</p><p><strong>Conclusion: </strong>This case report emphasizes the importance of early recognition and treatment to prevent long-term complications, both pathological and iatrogenic.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"737-745"},"PeriodicalIF":0.6,"publicationDate":"2025-11-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659664/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647340","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-11-20eCollection Date: 2026-01-01DOI: 10.1159/000549674
Yousef Alfadhli, Reem Kermani, Mohammad Shehab
Introduction: Crohn's disease (CD) is a chronic, relapsing-remitting disorder affecting the gastrointestinal tract that falls under the umbrella term of inflammatory bowel disease (IBD). Its etiology is unknown, but it is suggested that there is a strong genetic and environmental interaction, leading to its development. The mainstay of treatment for patients with moderate to severe CD is advanced therapy, such as biologics or small molecules. Despite the use of these medications, the disease may be unresponsive to these agents, leading to refractory disease. The use of novel therapies expanded the treatment options for patients with refractory CD or those who failed multiple biologics. Upadacitinib (UPA), a small molecule drug, belongs to a class of medications known as Janus kinase (JAK) inhibitors. UPA is known to be effective against CD with a dosage of either 15 or 30 mg. However, this dose deemed to be ineffective in the cases we reported. A higher maintenance dose of 45 mg was introduced and the patient's reported resolution of symptoms. It is important to note that larger studies are required to prove the efficacy of this treatment for CD.
Case presentations: This is a case-series of 3 patients with a history of refractory CD and prior exposure to at least 1 biologic agent, who achieved clinical remission and endoscopic healing on UPA maintenance therapy 45 mg orally once daily.
Conclusion: This case series illustrates potential benefit and supports the rationale for larger controlled studies evaluating high-dose UPA maintenance therapy.
{"title":"High-Dose Upadacitinib Maintenance Therapy for Refractory Crohn's Disease: A Case Series and Review of the Literature.","authors":"Yousef Alfadhli, Reem Kermani, Mohammad Shehab","doi":"10.1159/000549674","DOIUrl":"10.1159/000549674","url":null,"abstract":"<p><strong>Introduction: </strong>Crohn's disease (CD) is a chronic, relapsing-remitting disorder affecting the gastrointestinal tract that falls under the umbrella term of inflammatory bowel disease (IBD). Its etiology is unknown, but it is suggested that there is a strong genetic and environmental interaction, leading to its development. The mainstay of treatment for patients with moderate to severe CD is advanced therapy, such as biologics or small molecules. Despite the use of these medications, the disease may be unresponsive to these agents, leading to refractory disease. The use of novel therapies expanded the treatment options for patients with refractory CD or those who failed multiple biologics. Upadacitinib (UPA), a small molecule drug, belongs to a class of medications known as Janus kinase (JAK) inhibitors. UPA is known to be effective against CD with a dosage of either 15 or 30 mg. However, this dose deemed to be ineffective in the cases we reported. A higher maintenance dose of 45 mg was introduced and the patient's reported resolution of symptoms. It is important to note that larger studies are required to prove the efficacy of this treatment for CD.</p><p><strong>Case presentations: </strong>This is a case-series of 3 patients with a history of refractory CD and prior exposure to at least 1 biologic agent, who achieved clinical remission and endoscopic healing on UPA maintenance therapy 45 mg orally once daily.</p><p><strong>Conclusion: </strong>This case series illustrates potential benefit and supports the rationale for larger controlled studies evaluating high-dose UPA maintenance therapy.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"20 1","pages":"1-7"},"PeriodicalIF":0.6,"publicationDate":"2025-11-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12721722/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145818113","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-11-19eCollection Date: 2025-01-01DOI: 10.1159/000549673
Joseph L Maniaci, Allison J Burbank, Evan S Dellon
Introduction: Non-response to treatment in eosinophilic esophagitis (EoE) is common, and the approach to treatment-refractory patients with EoE is challenging. With increasing knowledge of EoE pathogenesis, mechanisms can be targeted by medications that are approved for other atopic diseases with a similar pathophysiology.
Case presentation: We present an adult with EoE who had longstanding symptoms prior to diagnosis, developed severe fibrostenosis with the narrow caliber phenotype requiring multiple esophageal dilations, and was previously nonresponsive to proton pump inhibitors (PPIs), topical corticosteroids (tCS), and food elimination diets (FED). Because of his concomitant severe asthma, he was treated with the eosinophil-depleting biologics mepolizumab and benralizumab. These reduced eosinophil counts on esophageal biopsy but did not improve symptoms, endoscopic features, or other histologic findings. He continued to require esophageal dilation for symptom control. His asthma control also remained suboptimal, prompting a trial of tezepelumab, an antibody against thymic stromal lymphopoietin (TSLP). In addition to controlling asthma, he improved across EoE symptom, endoscopic, and histologic metrics over a year of follow-up, including improvements in the EoE Endoscopic Reference Score, esophageal caliber, basal zone hyperplasia, dilated intercellular spaces, and lamina propria fibrosis as measured by the EoE Histologic Scoring System.
Conclusion: This patient, who did not respond to PPI, FED, tCS, mepolizumab, or benralizumab, ultimately had response to tezepelumab across multiple outcome domains of EoE. This demonstrates proof-of-concept that targeting TSLP may be a promising modality in EoE and supports further study of this mechanism for treatment of EoE.
{"title":"Successful Treatment with Tezepelumab for an Adult with Refractory and Severe Fibrostenotic Eosinophilia Esophagitis: A Case Report.","authors":"Joseph L Maniaci, Allison J Burbank, Evan S Dellon","doi":"10.1159/000549673","DOIUrl":"10.1159/000549673","url":null,"abstract":"<p><strong>Introduction: </strong>Non-response to treatment in eosinophilic esophagitis (EoE) is common, and the approach to treatment-refractory patients with EoE is challenging. With increasing knowledge of EoE pathogenesis, mechanisms can be targeted by medications that are approved for other atopic diseases with a similar pathophysiology.</p><p><strong>Case presentation: </strong>We present an adult with EoE who had longstanding symptoms prior to diagnosis, developed severe fibrostenosis with the narrow caliber phenotype requiring multiple esophageal dilations, and was previously nonresponsive to proton pump inhibitors (PPIs), topical corticosteroids (tCS), and food elimination diets (FED). Because of his concomitant severe asthma, he was treated with the eosinophil-depleting biologics mepolizumab and benralizumab. These reduced eosinophil counts on esophageal biopsy but did not improve symptoms, endoscopic features, or other histologic findings. He continued to require esophageal dilation for symptom control. His asthma control also remained suboptimal, prompting a trial of tezepelumab, an antibody against thymic stromal lymphopoietin (TSLP). In addition to controlling asthma, he improved across EoE symptom, endoscopic, and histologic metrics over a year of follow-up, including improvements in the EoE Endoscopic Reference Score, esophageal caliber, basal zone hyperplasia, dilated intercellular spaces, and lamina propria fibrosis as measured by the EoE Histologic Scoring System.</p><p><strong>Conclusion: </strong>This patient, who did not respond to PPI, FED, tCS, mepolizumab, or benralizumab, ultimately had response to tezepelumab across multiple outcome domains of EoE. This demonstrates proof-of-concept that targeting TSLP may be a promising modality in EoE and supports further study of this mechanism for treatment of EoE.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"784-793"},"PeriodicalIF":0.6,"publicationDate":"2025-11-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12707971/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145773626","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}