Case Reports in Gastroenterology最新文献

Introduction: Perivascular epithelioid cell tumor (PEComa) is a rare neoplastic mesenchymal tumor, more frequently found in the uterus, although it can occur in different organs. Hepatic PEComa is extremely rare, with only a few cases described in the literature.

Case presentation: We present a case report of a 33-year-old female patient with a history of macroprolactinoma. She was initially referred to our Department due to a 9-mm hepatic nodule incidentally diagnosed in an abdominal ultrasound in 2018. She was asymptomatic. Follow-up ultrasound showed a growth from 9 mm to 16 mm in 2019 and 30 mm in a liver magnetic resonance imaging (MRI) scan in 2022. The case was discussed in a multidisciplinary team meeting, and since malignant transformation or hepatocellular carcinoma could not be ruled out, the decision was to undergo hepatic resection. An open hepatic subsegmentectomy of segment 5 was performed, with uneventful postoperative period. The definitive diagnosis was hepatic PEComa.

Conclusion: Hepatic PEComas are rare liver tumors, and their preoperative diagnosis is challenging due to the lack of specific radiological features. In most cases, the diagnosis is only confirmed through histopathological and immunohistochemical studies. Resection of the lesion appears to be the curative treatment; however, due to the rarity of the condition, there are no studies comparing surgical treatment with other options. In our case, the hypervascular lesion was initially misdiagnosed as an adenoma. PEComas should be considered as a differential diagnosis in liver nodules with well-defined margins and increased uptake in the arterial phase in computed tomography or MRI scan. Surgical resection was curative, and no recurrence was detected during the patient's follow-up.

Introduction: Although cases of severe pancreatitis causing fistula formation into the colon have been documented, the reverse process of colitis causing a pancreatic fistula remains undocumented.

Case presentation: We present the case of a 79-year-old male with severe colitis resulting in perforation and pericolonic abscess formation adjacent to the pancreas, which resulted in an internal pancreatic fistula and pancreatic ascites. After 2 paracenteses, our patient ultimately underwent endoscopic retrograde cholangiopancreatography (ERCP) with sphincterotomy and pancreatic duct stent placement. The patient clinically improved and was ultimately discharged.

Conclusion: Follow-up ERCP was performed 2 months after discharge and showed no contrast extravasation, illustrating closure of the previous pancreatic fistula. Ultimately, our case demonstrates that cases of severe colitis may contribute to adjacent pancreatic fistula and ascites formation.

Introduction: Tonsillectomy is a safe and common procedure. It has not been associated with the development of hypercontractile conditions of the esophagus. Jackhammer esophagus is a type of hypercontractile esophagus with vigorous contraction of the esophagus associated with dysphagia and chest pain.

Case presentation: A 42-year-old woman with no previous history of gastroesophageal dysfunction or symptoms developed Jackhammer esophagus with severe dysphagia and chest pain after undergoing tonsillectomy and adenoidectomy.

Conclusion: Although Jackhammer esophagus has been reported as a complication of several invasive procedures, this is the first report of Jackhammer esophagus as a complication of tonsillectomy and adenoidectomy.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: The narrow time frame in which UC and GNET were diagnosed warrants further investigation into their potential relationship.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.

Introduction: 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.

Case presentation: 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.

Conclusion: 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.