Case Reports in Gastroenterology最新文献

Antimitochondrial antibody-positive primary biliary cholangitis (AMA-pos PBC) is an autoimmune disorder in which monoclonal antibodies are produced against epitopes in the mitochondrial membranes of biliary epithelial cells, resulting in progressive nonsuppurative biliary cholangitis. Up to 5% of patients lack these autoantibodies, termed antimitochondrial antibody-negative (AMA-neg) PBC. Although a somewhat new variant of AMA-pos PBC, it is not an overlapping syndrome. Few studies to date have described this phenomenon. An 87-year-old woman was referred to our clinic with elevated serum alkaline phosphatase (714 U/L). She reported fatigue but no other symptoms. A physical examination revealed a benign lesion and bilateral lower extremity swelling secondary to lymphedema. The serological profile was significant for a high antinuclear antibody titer (>1:2,560) with a centromere pattern and negative for antimitochondrial antibody (AMA). The hepatitis panel was negative for viruses A, B, and C. Her serum immunoglobulin G level was 871 mg/dL (normal, <1,600 mg/dL). The rest of the serological tests, including anti-smooth muscle antibodies (ASMA) and anti-liver/kidney microsomal antibodies, were negative. Computed tomography of the abdomen and pelvis without contrast showed normal liver parenchyma and no acute intra-abdominal pathology. Histopathology indicated florid duct lesions. The background parenchyma showed no significant steatosis, and inflammatory changes were limited to the portal areas. Periodic acid-Schiff staining revealed intact hepatic parenchyma and architecture. The patient was diagnosed with AMA-neg PBC and responded well to ursodeoxycholic acid therapy. This case highlights the importance of recognizing AMA-neg PBC as a variant of AMA-pos PBC and differentiating between them. Autoimmune cholangitis is a vague and imprecise condition. All patients with AMA-negative PBC should be tested for other PBC-specific autoantibodies. Although the prognosis and bile duct damage and loss are worse in AMA-neg PBC for unknown reasons, treatment remains the same for both.

The diagnosis of chronic mesenteric ischaemia is typically based on angiographic findings along with a classic symptomatology. Only a few methods are available for functional testing to establish the diagnosis, such as indirect measurement of the splanchnic blood flow and hepatic vein oxygenation. The present case is a 76-year-old woman with weight loss and intermittent abdominal pain who was clinically suspected of chronic mesenteric ischaemia based on severe stenosis of the celiac artery and superior mesenteric artery. Measurement of the total splanchnic blood flow and hepatic vein oxygenation, however, showed a normal perfusion after meal stimulation, as well as an increased hepatic vein oxygenation, indicating normal flow reserves. This was likely due to a richly developed Riolan's anastomosis arising from the inferior mesenteric artery. The present case advocates the need for functional testing when diagnosing chronic mesenteric ischaemia.

Oral lichen planus (OLP), a chronic inflammatory mucocutaneous disease, is an extrahepatic manifestation of a hepatitis C virus infection. In recent years, direct-acting antivirals (DAAs) have made great strides in the treatment of hepatitis C. However, there might be a lack of information about the treatment strategies available among those with this condition. Herein, we report a case of an 85-year-old female patient who was diagnosed with hepatitis C at the age of 55 but had not received antiviral treatment over the past 30 years. She underwent DAA treatment following a recommendation from her oral surgeon after the onset of OLP. The patient had declined interferon therapy in the past, owing to concerns about its side effects. She was unaware of the benefits of DAA treatment, probably due to communication difficulties caused by senile hearing loss. Consultation with an oral surgeon for an erosive form of OLP led her to receive antiviral therapy for hepatitis C. She achieved a sustained virologic response (SVR) following the DAA treatment, along with improvements in the signs and symptoms of OLP. Oral surgeons play an important role as gatekeepers in guiding untreated hepatitis patients toward appropriate treatment.

Acute gastrointestinal bleeding (AGIB) is a common condition leading to hospitalization and is associated with significant morbidity and mortality. Various endoscopic treatments have been reported for AGIB, while its endoscopic hemostasis treatment by combined modality with PuraStat and endoscopic hemoclips remains less well documented. We report 6 cases of AGIB, i.e., 5 cases of ulcer bleeding and 1 case of ruptured gastric varices in 3 males and 3 females aged 49-97 years (mean age, 77 years), 2 and 1 of whom had been on antithrombotic drugs and nonsteroidal anti-inflammatory drugs, respectively, with 2 of these found to be in critical condition with hemorrhagic shock. Types of bleeding treated included oozing bleeding from visible vessels (n = 3), spurting bleeding from visible vessels (n = 2) and from gastric varices (n = 1). In all cases, complete hemostasis was achieved with no rebleeding. To our knowledge, this report represents a valuable addition to the AGIB literature describing endoscopic hemostasis by combined modality therapy with PuraStat and endoscopic hemoclips.

The diagnosis of carcinoid heart disease as a cause of ascites can be hard to establish. We report a patient with well-differentiated neuroendocrine neoplasm of the liver who presented with high serum ascites albumin gradient and high protein ascites due to carcinoid heart disease (CHD). As ascites caused by CHD are rare, the etiology can easily be overlooked, especially in the setting of alcohol use disorder and portal hypertension. Through our case report, we emphasize the importance of physical examination and peritoneal fluid analysis in the diagnosis of CHD. As the management of CHD requires a multidisciplinary approach, early diagnosis is crucial so that relevant specialists can have the opportunity for early intervention in order to produce the best patient outcome.

We report a case of a 70-year-old male with delayed perforation in the cecum treated by endoscopic ultrasonography-guided drainage for a pelvic abscess. The lesion was a 50-mm laterally spreading tumor, and endoscopic submucosal dissection (ESD) was performed. No perforation was detected during the operation, and en bloc resection was achieved. He had fever and abdominal pain on postoperative day (POD) 2. Computed tomography (CT) revealed the intra-abdominal free air, leading to a diagnosis of delayed perforation after ESD. Vital signs were stable, the perforation was considered minor, and endoscopic closure was attempted. The colonoscopy under fluoroscopy showed no perforation in the ulcer and no leakage of the contrast medium. He was managed conservatively with antibiotics and nothing per os. Symptoms improved; however, a follow-up CT on POD 13 revealed a 65-mm pelvic abscess, and endoscopic ultrasound (EUS)-guided drainage was successfully performed. The follow-up CT on POD 23 showed the reduction of abscess, and the drainage tubes were removed. Emergent surgical treatment is crucial in delayed perforation because it has a poor prognosis, and reports of conservative therapy for colonic ESD with delayed perforation are few. The present case was managed with antibiotics and EUS-guided drainage. Thus, EUS-guided drainage can be a treatment option for delayed perforation after colorectal ESD, if the abscess is localized.

Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare systemic vasculitis of small to medium vessels. Gastrointestinal involvement is uncommon and is associated with higher mortality. Treatment is based on empiric evidence. In this article, we report a case of EGPA-related pancolitis and stricturing small bowel disease managed with a combination of mepolizumab and surgical resection.

Leukocytoclastic vasculitis (LCV) is rarely associated with anti-tumor necrosis factor [TNF] α therapy. We report a 22-year-old man with new onset of a pustular rash on his bilateral upper and lower extremities while on adalimumab therapy for Crohn's disease. Skin biopsy of the affected area showed perivascular extravasation of erythrocytes, neutrophils, eosinophils and vascular damage surrounding blood vessels associated with fibrin, consistent with LCV. Patient was treated with topical steroids and subsequently transitioned to ustekinumab therapy with follow-up colonoscopy showing minimal active disease. Our report highlights the association of a unique dermatologic autoimmune manifestation with TNF-targeted therapy in a patient with Crohn's disease.

Zellweger spectrum disorders (ZSDs) are known to present with variable hepatic manifestations ranging from benign hepatosplenomegaly and elevated liver enzymes to advanced liver cirrhosis with hepatocellular carcinoma. However, the progression of liver disease in ZSD patients over time is poorly characterized due to scarcity of the disease. Herein, we report a case of newly diagnosed liver cirrhosis in a ZSD patient with rapid progression and fatal outcome to demonstrate key clinical learning points.

Retroperitoneal and mediastinal emphysema after colon resection is extremely rare, especially in the absence of anastomotic leakage. The feasibility and safety of conservative treatment for this complication are unknown. We report a patient who underwent open sigmoid colon resection for colon cancer and developed retroperitoneal and mediastinal emphysema that was not caused by anastomotic leakage. Retroperitoneal and mediastinal emphysema occurred as a result of diverticular perforation. We were able to treat this patient successfully with conservative management.