Case Reports in Gastroenterology最新文献

Introduction: Endoscopic clot removal in complex huge esophageal hematoma is a rare clinical entity.

Case presentation: A 48-year-old male patient presented to our hospital with vomiting blood with black stool for 1 day after drinking alcohol. Esophageal hematoma was diagnosed after chest CT and EGD. After conservative treatment with intravenous fluids, the patient's symptoms of vomiting blood persisted and the hemoglobin decreased significantly. We decided to perform another EGD: the huge hematoma had ruptured. We performed endoscopic hemostasis and blood clot removal. Long-term postoperative follow-up shows the mucosa heals well.

Conclusion: Endoscopic clot removal is an effective method of treating esophageal huge hematoma accompanied by rupture.

Introduction: Intramural oesophageal dissection is a rare oesophageal injury that occurs primarily in the elderly and is associated with aggravating factors such as coagulopathy or anticoagulation therapy. It can be emetogenic, trauma-related, iatrogenic, or spontaneous.

Case presentation: We present an unusual case of an 87-year-old woman on low dose aspirin who presented with severe chest pain and sudden onset of haematemesis without prior history of forceful vomiting or Valsalva manoeuvres. An emergency upper gastrointestinal endoscopy confirmed the initial diagnosis of a spontaneous intramural oesophageal haematoma and a follow up computed tomography scan of the chest confirmed the presence of an intramural oesophageal dissection.

Conclusion: Spontaneous intramural oesophageal dissections should be considered as a differential diagnosis in a patient presenting with acute chest pain without known risk factors such as coagulopathy.

Introduction: Collagenous sprue (CS) is an extremely rare enteropathy of unknown etiology and if untreated, can lead to substantial morbidity and mortality. CS often copresents with celiac disease. The presence of a subepithelial collagen band on histology differentiates CS from celiac disease, as both have villous blunting. The management of the two diseases is different, and thus it is critical that the proper diagnosis of CS is made promptly to prevent complications.

Case presentation: We present a case report of CS in an elderly male who was initially diagnosed with celiac disease alone before returning to care years later with unresolved gastrointestinal symptoms.

Conclusion: Clinicians must verify that CS has been ruled out following a celiac disease diagnosis. In regard to CS's mechanism, CS's high frequency of comorbid autoimmune conditions and its robust response to corticosteroids support an immune-mediated process. Future research should continue to aim to elucidate the mechanism as it would allow for a more targeted approach to treatment, such as anti-fibrotic or specific immunomodulator therapy.

Introduction: Organ-preserving treatment for rectal cancer using local excision (LE) and/or chemoradiotherapy (CRT) is increasingly used. Locoregional metastasis precludes LE and locoregional regrowth, recurrence, or persistence after LE or chemoradiation (CRT) may prompt total mesorectal excision (TME). We believe that the time has passed to make such life-changing treatment decisions without pathological confirmation and investigated the use of linear endoscopic ultrasound with fine-needle aspiration (EUS-FNA).

Case presentations: We report 8 cases of suspected locoregional tumor growth (LRTG) on MRI: adjacent or in the rectal wall, within the mesorectal fascia, high presacral region, and obturator foramen. MRI images were studied thoroughly before and during EUS to identify the target lesion using rectal EUS-FNA. Patients were prepared using an enema. The procedure was performed on an outpatient basis without conscious sedation. FNA was performed using a 25G needle. The patient received a 3-day course of ciprofloxacin after the procedure to prevent infection of the perirectal space. Identification of the target was the most difficult part of EUS but was successful in all cases. FNA revealed adenocarcinoma in 7 cases. Five cases were confirmed by TME results: 1 patient died before the operation, and 1 patient was treated with CRT. One patient with a suspected node in the obturator foramen was free of tumors on FNA. The TME resection specimen contained 31 lymph nodes without metastasis. All procedures were well tolerated, and no complications were observed.

Conclusion: Suspected LRTG on MRI can be confirmed using EUS-FNA. In the era of organ-preserving treatment for rectal cancer, EUS-FNA may play a supportive role when considering TME or CRT.

Introduction: Cryptogenic multifocal ulcerative stenosing enteritis (CMUSE) is a rare and underrecognized small bowel disorder that often mimics Crohn's disease, leading to delays in diagnosis and misdirected treatment. Given its relapsing nature and resistance to conventional inflammatory bowel disease (IBD) therapies, CMUSE presents significant diagnostic and therapeutic challenges.

Case presentation: We present the case of a 41-year-old male with chronic anemia, fatigue, weight loss, and intermittent abdominal pain with melena, who remained undiagnosed for 9 years despite extensive evaluations. Imaging and endoscopy failed to identify a definitive cause, and management with TNF inhibitors and IL-12/IL-23 blockade provided only temporary relief. The patient required multiple surgical resections due to recurrent strictures. Pathological examination consistently revealed multifocal jejunal ulceration with stenosis but lacked granulomas, vasculitis, or systemic inflammatory markers, ultimately confirming CMUSE. Given its distinct pathology and treatment resistance, differentiating CMUSE from Crohn's disease is essential. The patient's ongoing management includes upadacitinib, a JAK1 inhibitor, which may help modulate immune pathways contributing to ulcer formation and stricture development.

Conclusion: This case underscores the need for heightened clinical recognition of CMUSE, particularly in patients with unexplained small bowel strictures and ulceration unresponsive to standard IBD therapies. Genetic testing may aid in distinguishing CMUSE from Crohn's disease, preventing unnecessary immunosuppressive treatments. Further research is necessary to establish effective, targeted therapies and improve outcomes for patients with this rare condition.

Introduction: Bronchoesophageal fistulas (BOF) have predominantly been found to result in a decreased quality of life and an increased rate of mortality, particularly due to their severe complications and difficult treatment.

Case presentation: This report discusses the case of a 71-year-old female who presented with shortness of breath and a continuous cough secondary to a fistula between the oesophagus and right bronchus on the background of squamous cell carcinoma (SCC). This patient was seen by oncologists for the treatment of her lung cancer after right middle and lower lobe lobotomies, which was then treated with radiotherapy. On admission, a computed tomography scan revealed that the patient had a BOF due to therapeutic radiotherapy for SCC. She underwent intervention from the gastrointestinal and respiratory physicians to treat the BOF. Oesophageal stent placement was performed for treatment; however, the BOF remained patent, so a bronchial stent was considered for insertion. While the patient was awaiting the bronchial stent, she died.

Conclusion: This case highlights the complexities and challenges of BOFs, emphasising the need for further research and documentation to improve treatment strategies. More studies are needed to determine when oesophageal stenting is preferred over bronchial stenting and to evaluate the suitability and safety of dual stenting in both the oesophagus and bronchus.

Introduction: Benign recurrent intrahepatic cholestasis (BRIC) is a rare autosomal recessive inherited disorder characterized by intermittent jaundice episodes.

Case presentation: We report a 25-year-old male patient with numerous hospitalizations for jaundice. The diagnosis of BRIC type 1 was established by liver biopsy, genetic analysis, and the exclusion of alternative etiologies of cholestasis. A novel missense heterozygous variant was identified in the ATP8B1 gene (c.2081T>A). The acute cholestatic attack, along with associated complications such as distal renal tubular acidosis and acute pancreatitis, was successfully managed with plasmapheresis and rifampicin.

Conclusion: Novel mutations that differ from those documented in the literature in combination with renal tubular acidosis may enhance our comprehension of this topic.

Introduction: The lumen-apposing metal stent (LAMS) is a novel prosthesis for fistula formation between the gastrointestinal and cyst walls. Bleeding complications occur in 10% of cases mostly during LAMS placement. We present a case of recurrent bleeding following LAMS placement.

Case presentation: A 47-year-old male was admitted to a local hospital for acute pancreatitis and subsequently referred for endoscopic drainage because of an enlarging pseudocyst near the tail of the pancreas. Treatment involved LAMS and endoscopic ultrasound-guided transmural drainage. Posttreatment computed tomography revealed cyst shrinkage; however, upper gastrointestinal bleeding was noted after discharge. Endoscopy identified bleeding within the fistula, which was controlled by spraying an absorbable local hemostatic agent into the cavity. After ensuring the absence of recurrent bleeding for approximately 1 month, the LAMS was removed under endoscopy. Following LAMS removal, arterial bleeding was observed within the cavity, prompting an emergency angiography. Angiography revealed bleeding from a pseudoaneurysm of the splenic artery, which was treated with coil embolization. No rebleeding occurred after the procedure.

Conclusion: Bleeding 1 week after placement or during removal is rare. However, following LAMS placement, there is a risk of bleeding until removal, with arterial bleeding often occurring at the time of removal. Therefore, it is necessary to establish a system that allows for prompt vascular embolization treatment.

Introduction: Zollinger-Ellison syndrome (ZES) is a rare entity consisting of tumors called gastrinomas in the stomach, pancreas, and duodenum. It usually presents with symptoms of acid hyper secretion including abdominal pain, diarrhea, nausea, and vomiting and can be misdiagnosed as peptic ulcer disease, hiatal hernia, and gastroesophageal reflux disease.

Case presentation: We report a case of ZES presenting duodenal perforation and later mimicking a hiatal hernia. This case highlights a complex presentation and underscores the importance of thorough evaluation, multidisciplinary management, and including rare diagnosis in the differential. Our patient presented to the hospital with nausea, back pain, and abdominal pain, and imaging demonstrated a perforated duodenum which was managed with surgical repair. Following surgery, the patient continued to have worsening nausea and acid reflux which was deemed to be due to a hiatal hernia noted on prior imaging until an esophagogastroduodenoscopy (EGD) was performed which confirmed the diagnosis of ZES.

Conclusion: Our case emphasizes the importance and necessity of doing an EGD in a timely fashion to ensure that a diagnosis of rare ZES is not missed.

Introduction: The medical treatment of refractory collagenous colitis with a concomitant symptomatic lymphocytic disorder of the upper gastrointestinal tract is very challenging with scarce evidence.

Case presentation: We present a 61-year-old female patient with a long-standing highly refractory collagenous colitis with a concomitant symptomatic lymphocytic disorder with villous atrophy and intraepithelial lymphocytes of the upper gastrointestinal tract causing severe watery diarrhoea with severe hypokalemia and recurrent episodes of prerenal kidney injuries requiring several hospital admissions. Celiac serology as well as genetic analyses (HLA-DQ2/DQ8) were negative, and other common etiologies of intraepithelial lymphocytosis and villous atrophy were ruled out. Considering the similar course of the disease in the upper and lower gastrointestinal tract for a time period of more than 20 years, a common etiologic relationship, particularly an autoimmune disorder seems to be very likely in this patient. Several therapies such as budesonide, immunomodulators, and the biologics infliximab and vedolizumab had to be stopped due to either non-response, loss-of-response or drug-related side effects. However, the patient responded immediately to the JAK-1 inhibitor upadacitinib, with documented remission for more than 1 year.

Conclusion: For the first time, a prompt and significant response to upadacitinib in a patient with refractory collagenous colitis with upper gastrointestinal tract involvement was shown, suggesting upadacitinib as therapy of choice in severe therapy-refractory cases of collagenous colitis, particularly with concomitant upper gastrointestinal tract involvement.