Case Reports in Gastroenterology最新文献

Extracolonic manifestations of Clostridium difficile have been rarely reported. We herein report a case of a 60-year-old immunocompetent man presenting with fever, nausea, abdominal pain, and loose stools for 2 weeks. Triple-phase liver computed tomography demonstrated pyogenic liver abscesses and portal pylephlebitis. Blood cultures grew C. difficile and Bacteroides fragilis, and liver abscess cultures grew Proteus mirabilis, Escherichia coli, and the viridans group Streptococci. Antibiotics coverage was selected to direct at all identified organisms. This demonstrates an unusual case of C. difficile bacteremia in a patient with polymicrobial pyogenic liver abscesses and pylephlebitis.

The patient was a woman in her 40s who was diagnosed with Crohn's disease (CD) of the large and small intestines in 1996. In 2005, she was referred to our hospital for treatment. We treated her for 17 years with corticosteroids, biologics, immunosuppressive agents, 5-aminosalicylic acid, and nutrition care. However, her Crohn's Disease Activity Index remained between 200 and 250, indicating refractory CD. During her medical treatment, the patient also underwent 3 operations. One year ago, the patient became pregnant through in vitro fertilization. Even after pregnancy was confirmed, the patient continued her treatment for refractory CD with ustekinumab, granulocyte apheresis, and budesonide. Nonetheless, her CD was highly active during pregnancy, and she experienced various complications: sigmoid volvulus at gestational week 15, venous thrombosis at gestational week 17, nontraumatic rib fracture due to fetal movement at gestational week 32, and sepsis from central venous catheter infection at gestational week 37. At gestational week 38, the patient gave birth by emergency cesarian delivery. This paper reports details of the case in which delivery was achieved after various complications were overcome and discusses previous relevant reports.

Eosinophilic granulomatosis with polyangiitis (EGPA), historically named Churg-Strauss syndrome, is a rare vasculitis affecting small- and medium-sized blood vessels. The disease has a predilection for numerous organs including the lungs, sinuses, kidneys, heart, nerves, and gastrointestinal tract but is prominently associated with asthma, rhinosinusitis, and eosinophilia. Gastrointestinal involvement is common; however, a gastrointestinal manifestation as the cardinal symptom following an infection is atypical. Here, we present a case of a 61-year-old male who presented with persistent diarrhea following a toxigenic Clostridium difficile infection despite multiple antibiotic courses. Repeat testing confirmed eradication of the infection, and further evaluation with colon biopsy revealed small and medium-sized vasculitis with eosinophilic infiltration and granulomas. Treatment with prednisone and cyclophosphamide resulted in rapid improvement of his diarrhea. Gastrointestinal symptoms in EGPA are associated with worse prognosis, so prompt identification and treatment of the disease is crucial. EGPA is rarely documented in histopathological samples from the gastrointestinal tract as endoscopic biopsies are typically too superficial to sample the submucosal layer containing the affected vessels. Additionally, the link between EGPA and infections as a potential trigger has not been clearly established, but gastrointestinal EGPA manifesting after a colonic infection raises concerns that this may have been a triggering event. Ultimately, further study is needed to understand, diagnose, and treat gastrointestinal and postinfection EGPA.

We treated a 39-year-old Japanese man who was admitted for an abdominal mass. He had had neurofibroma-like skin lesions since childhood. Computed tomography and endoscopic ultrasound results were consistent with a tumor in the small intestine. Although the tumor was undetectable by single-balloon endoscopy, the patient's background and imaging results led us to suspect a gastrointestinal stromal tumor (GIST). He also met the diagnostic criteria for neurofibroma type 1 (NF1). We performed a surgical removal of the tumor, and the biopsy results led to a definitive diagnosis of GIST. Small bowel GISTs should be considered in cases of NF1.

Colonic metastasis from ovarian cancer is extremely rare, with only seven reported cases. A 77-year-old woman who had previously undergone surgery for ovarian cancer was admitted to a local hospital with anal bleeding. Histopathological analysis confirmed the presence of adenocarcinoma. Colonoscopy revealed a descending colon tumor. The patient was diagnosed with Union for International Cancer Control T3N0M0 descending colon cancer or colon metastasis of the ovarian cancer. Laparoscopic left colectomy was performed; intraoperative frozen section diagnosis confirmed metastasis from ovarian cancer, and the absence of invasion to the serosal surface suggested hematogenous metastasis. This is the first case of colonic metastasis from ovarian cancer that was diagnosed using an intraoperative frozen section and laparoscopically treated.

Cowden syndrome is characterized by several clinical features related to tumorous lesions primarily consisting of systemic hamartomas. The mutation of a tumor suppressor gene, the PTEN gene, is etiologically involved. As gastrointestinal lesions, polyps of all digestive tracts involving the esophagus to rectum develop. In patients with Cowden syndrome, the risk of colorectal cancer may increase. However, the characteristics of colorectal cancer in these patients remain to be clarified and sufficient findings regarding chemotherapy have not been obtained. A 39-year-old man was treated with a colonic stent for colitis obstructive due to circumferential transverse colon carcinoma. After decompression, elective extended laparoscopic right hemicolectomy was performed. Preoperative systemic detailed examination revealed characteristic dermal/mucosal findings, polyposis of the upper digestive tract, and a thyroid tumor. On PTEN gene sequencing, a mutation was detected at codon 130 of exon 5, leading to a diagnosis of Cowden syndrome. Postoperative adjuvant chemotherapy was performed for 6 months, but recurrent peritoneal dissemination was observed 1 month after its completion. FOLFOXIRI + bevacizumab therapy was started. Transiently, a partial response was achieved in peritoneally disseminated nodes according to the RECIST. There was no increase in the volume of cancerous ascites. However, an increase in the volume of ascites and local relapse were noted at the completion of the tenth course. The regimen was switched to FOLFIRI + panitumumab, but peritoneal dissemination exacerbated and the patient died 18 months after surgery.

When the etiology of pancreatitis cannot be determined despite sufficient investigation, recurrence and progression to chronic pancreatitis often involve genetic mutations. Herein, we describe a case of recurrent pancreatitis with the IVS3+2T>C mutation in the serine protease inhibitor Kazal type 1 (SPINK1) gene that progressed to chronic pancreatitis in only 3 years. A 35-year-old man was referred to our hospital, where he was diagnosed with mild pancreatitis and was treated conservatively. However, the patient experienced recurrent episodes of pancreatitis, which progressed to become chronic pancreatitis with a pancreatic calcification 1 year later. After 3 years, the patient developed pancreatic duct stenosis and required a pancreatic duct stent placement. Regarding the cause of chronic pancreatitis, alcohol abuse was ruled out based on history taking. Considering the course of treatment, autoimmune pancreatitis and obstructive pancreatitis, such as pancreatic divisum, were also ruled out. Finally, a germline genetic test was performed to determine the etiology of pancreatitis, which revealed the IVS3+2T>C mutation in SPINK1. This case shows the importance of genetic testing in patients with idiopathic pancreatitis to determine their etiology and is a rare incident that can report the progression of the disease from acute to chronic pancreatitis.

Tyrosine kinase inhibitors (TKIs) such as imatinib improve the prognosis of patients with gastrointestinal stromal tumors (GISTs). However, treatment options for GISTs are still limited, and the continuation of TKIs is difficult due to adverse events in some cases. The effectiveness of low-dose imatinib is unclear. We report 2 cases to show effectiveness of low-dose imatinib in patients with adverse events. The first case is a male in his early 60s with a history of intestinal GIST resection who was diagnosed with recurrent GIST with peritoneal dissemination. He was started on low-dose imatinib (300 mg) because of a history of subconjunctival hemorrhage after receiving postoperative imatinib. Follow-up contrast-enhanced ultrasonography revealed that the tumors had shrunk in size and number after 2 months of treatment with 300-mg imatinib. He continued this treatment and showed partial response for 8 months. The second case is a female in her late 70s with rectal GIST who was treated with imatinib 400 mg. Due to a severe skin lesion, she changed her treatment to sunitinib 2 months after initiation. However, new metastasis in the liver was confirmed after 4 months of administration of sunitinib. She underwent surgical esection of the rectal tumor to reduce the volume. After the surgery, low-dose imatinib (300 mg) with oral steroids was adopted. Follow-up confirmed the absence of recurrence at the rectum and no increase in hepatic tumor size for 18 months. Aggressive treatment with low-dose imatinib instead of discontinuation or alteration of treatment may benefit patients with unresectable and postoperative GISTs with sensible mutation to imatinib.

Granular cell tumor (GCT) was first described by Abrikossoff in 1926. It is a mostly benign tumor with rare malignant transformation. It is defined as a soft tissue neoplasm with abundant eosinophilic cytoplasm. The mean age of diagnosis for GCT is around 45 years. It is rare for GCT to be found in the gastrointestinal (GI) tract. Within the subset of GI tract, the colon is an extremely rare site for it to be found. Franburg-Smith histopathology criteria are used to differentiate a benign from a malignant GCT. The malignant form is aggressive with high recurrence rates after resection. Histopathology and immunohistochemical stains are used to make a definitive diagnosis. Herein, we present a rare case of an ascending colon polyp that was resected and found to be a benign GCT.

Hypereosinophilic syndrome (HES) is a rare condition characterized by hypereosinophilia in peripheral blood or tissue infiltrate and organ damage. HES has been associated with several diseases, including inflammatory bowel diseases (IBDs), especially ulcerative colitis (UC). In this report, we describe a case of a UC and primary sclerosing cholangitis patient who was diagnosed with HES and severe cardiovascular and neurological injury. During hospitalization, an extensive diagnostic workup was performed and secondary causes of hypereosinophilia were ruled out. The patient was treated with glucocorticoids and full anticoagulation with significant clinical improvement and a marked reduction in the eosinophil count. In the literature, hypereosinophilia in the IBD population has been related to the severity of the disease and worse prognosis. The high index of clinical suspicion and the accurate diagnosis of HES are essential to avoid delay in therapy and prevent complications.