Case Reports in Gastroenterology最新文献

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentations: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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 Escherichia coli 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.

Conclusion: 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.

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Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Discussion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: Filgotinib may be an effective second-line therapy in an emergency setting such as ASUC in patients unresponsive to conventional therapy.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

[This corrects the article DOI: 10.1159/000544108.].

Introduction: Primary (or idiopathic) panniculitis involving the intra-abdominal adipose tissue is rare, and its pathogenesis remains unknown. A case of primary eosinophilic panniculitis that involved the greater omentum of a girl is reported.

Case presentation: The patient, an 11-year-old girl, complained of dull periumbilical pain and nausea, and radiological examination showed a mass lesion in the abdomino-pelvic cavity. On laparoscopy, a plaque-like, flat mass was seen in the greater omentum, and laparoscopic omental resection was performed. On histopathological examination, the interlobular fibrous septa of omental adipose tissue were widened by inflammatory edema, prominent infiltration of eosinophils, and loose proliferation of myofibroblasts. Dense lymphocytic infiltration was also noted around small veins. Inflammatory changes were mild in the fat lobules, and fat necrosis and infiltration of lipid-laden macrophages were absent. Findings of obliterative phlebitis or arteritis were not seen.

Conclusion: Isolated involvement of the omentum by a panniculitic process is rare, and the pathogenesis of eosinophilic septal panniculitis found in the present case remains unknown, but involvement of a hypersensitivity reaction against some unknown stimuli is presumed, based on the histopathological resemblance of the omental lesions to erythema nodosum or eosinophilic panniculitis of the skin. We should keep in mind the possibility that the omental lesion in this patient is a harbinger of more serious immunological disorders. Careful, long-term follow-up and monitoring of the patient are needed.