Atypical, Levetiracetam-induced Hypersensitivity Syndrome Complicated by Fulminant Liver Failure in a Patient Undergoing Hemodialysis.

A 59-year-old man undergoing hemodialysis was administered levetiracetam, after which he developed a systemic rash, high fever, severe liver dysfunction, and leukocytopenia with reactivation of human herpes virus 6. Atypical drug-induced hypersensitivity (DIHS) was diagnosed, and prednisolone was administered at 60 mg/day. However, liver failure rapidly progressed, and the patient died 12 days following treatment. Despite the rarity of DIHS with concomitant fulminant liver failure from levetiracetam and sufficient clearance thereof by hemodialysis, our case suggests that this syndrome may still ensue, resulting in mortality, even in hemodialysis patients. Although no treatment has yet been established, strict monitoring and aggressive treatment may be required.

Low potency of fecal immunological surveillance testing soon after negative colonoscopy or resection of low-risk adenoma in average-risk patients.

BACKGROUND: Postcolonoscopy surveillance colonoscopy based on positive fecal occult blood testing (FOBT) is often performed, although its long-term efficacy has not been established. The aim of this study was to clarify the low potency of FOBT surveillance at short intervals after colonoscopy. METHODS: Colonoscopy was performed in 1308 average-risk patients, based on positive results of immunological FOBT [fecal immunological test (FIT)]. Patients were stratified according to the length of time since their last colonoscopy and their colonoscopy results [no adenoma or 1-2 small (<10 mm) adenomas]. Tumor detection rates were determined. RESULTS: The baseline patients characteristics did not differ between the groups. The advanced lesion detection rate (ALDR) among the patients who had never undergone a colonoscopy was 21.9% [95% confidence interval (CI), 19.1-25.0%]. Among the patients who had no adenoma detected in the previous colonoscopy within the past 5 years, the past 5-10 years and over 10 years, the ALDRs were 2.5% (95% CI, 1.0-5.5%), 4.1% (95% CI, 1.5-9.4%) and 9.3% (95% CI, 3.1-22.2%), respectively. Among the patients who had 1-2 small adenomas, the ALDRs were 7.4% (95% CI, 3.4-14.8%), 12.1% (95% CI, 4.2-27.9%) and 27.8% (95% CI, 12.2-51.2%), respectively. Invasive cancer was not observed in any patients within 5 years since the prior colonoscopy. CONCLUSION: In average-risk patients whose prior colonoscopy detected no adenomas or low-risk adenomas, postcolonoscopy surveillance by FIT has a low positive predictive value within a 5-year interval.

Intravascular Large B-cell Lymphoma Mimicking Hepatobiliary Infection: A Case Report and Literature Review.

Intravascular large B-cell lymphoma (IVLBCL) frequently involves the hepatobiliary system, but its clinical course and pathophysiology are still not fully known. We herein describe a case of IVLBCL mimicking acute hepatobiliary infection. An 85-year-old woman was admitted because of fever and epigastric pain, and she was diagnosed to have acute acalculous cholecystitis based on gallbladder wall thickening with fluid collection. The gallbladder swelling regressed within several days, and areas of intrahepatic hypoperfusion appeared. Inflammation continued despite treatment with antibiotics, and she died within 21 days. An autopsy examination revealed IVLBCL. IVLBCL can present as acute cholecystitis with an improvement in the imaging findings and the presence of a subsequent liver mass.

[Retroperitoneal Dedifferentiated Liposarcoma with Metaplastic Bone Formation : A Case Report and Review of the Literature].

A 69-year-old female was introduced to our department for a retroperitoneal mass, 25 mm in diameter, in the right perirenal space indicated by computed tomography (CT). On the basis of magnetic resonance imaging (MRI), retroperitoneal soft tissue sarcoma was suspected. Because she rejected surgical treatment, we continued imaging surveillance. Subsequently, the mass grew larger, but her intention did not change. At 21 months after initial consultation, CT revealed further increase of the above mass and a new lesion with calcification. Ultimately, she underwent mass resection with concomitant resection of the right kidney. Histological examination showed dedifferentiated liposarcoma with metaplastic bone formation and positive surgical margin, but she refused adjuvant chemotherapy. She has survived 7 months since the operation with no evidence of recurrence.

Large-cell neuroendocrine carcinoma arising from a gastritis cystica polyposa.

Gastritis cystica polyposa is a polypoid lesion that arises from the gastric mucosa at the gastrojejunal anastomotic site and is characterized by cystic dilation of the gastric glands. A 78-year-old man who underwent distal gastrectomy for a gastric ulcer with Billroth II reconstruction approximately 40 years previously, exhibited a gastritis cystica polyposa at the anastomotic site. Ulceration was observed on an annual endoscopic examination. Endoscopic ultrasonography revealed a submucosal hypoechoic mass with multiple cystic lesions. Gastrectomy was performed and histological examination revealed a large-cell neuroendocrine carcinoma with cystic dilation of the gastric glands. Here, we report the first case of a large-cell neuroendocrine carcinoma arising from a gastritis cystica polyposa. Endoscopic ultrasonography was effective at diagnosing a submucosal hypoechoic mass with cystic dilation of the gastric glands.

Early esophageal cancer with epidermization diagnosed and treated with endoscopic resection.

The patient was a 57-year-old man who had undergone endoscopic submucosal dissection for early esophageal cancer (distance from incisor tooth, 30 cm) when he was 50 years of age. Pathological findings showed squamous cell carcinoma invading the lamina muscularis mucosae and mild lymphatic invasion. Considering the possibility of lymph node metastasis and distant metastasis, we administered radiation chemotherapy (CDDP+ 5-FU, total radiation 41.4 Gy) in the same year. Two years later, follow-up endoscopy revealed a white, flat, elevated lesion in the thoracic esophagus (distance from incisor tooth, 36 cm) that was not stained by Lugol's iodine. A biopsy of this lesion was performed. Although esophageal epidermization was seen, there were no findings suggestive of malignancy. The lesion grew slightly during four and a half years of follow-up after identification. We performed a repeat biopsy of the lesion, and the tissue was diagnosed as atypical epithelium, so we performed endoscopic mucosal resection for diagnostic and therapeutic purposes. The postoperative pathological diagnosis was squamous cell carcinoma of T1a-LPM with epidermization due to its histological features. To the best of our knowledge, this is the first report of esophageal cancer accompanied by epidermization.

Intestinal-type gastric adenocarcinoma without Helicobacter pylori infection successfully treated with endoscopic submucosal dissection.

A 67-year-old woman was admitted to our hospital for further examination and for treatment of gastric neoplasia located on the posterior wall of the antrum of the stomach, as revealed by screening esophagogastroduodenoscopy. The patient had no history of Helicobacter pylori (H. pylori) eradication. Her serum H. pylori antibody and urea breath test results were negative, histopathological findings revealed no H. pylori bacteria, and endoscopic findings revealed no chronic gastritis. We performed endoscopic submucosal dissection (ESD). Histological examination of the resected tissues revealed the tumor to be composed of a well-differentiated tubular adenocarcinoma with a tubular-type adenoma confined to the mucosa. This adenocarcinoma exhibited immunohistochemical expression of CD10, MUC2, and Cdx2, but not MUC5AC or MUC6. This is an extremely rare case of H. pylori infection-negative, intestinal-type, differentiated gastric adenocarcinoma revealed by detailed immunohistochemical examination that was treated with ESD. The patient has had no recurrence of adenocarcinoma after ESD.

Non-Hodgkin's lymphoma accompanied by pulmonary involvement with diffuse ground-glass opacity on chest CT: a report of 2 cases.

We report 2 rare cases of non-Hodgkin's lymphoma accompanied by pulmonary involvement with diffuse ground-glass opacity. Histological examination of the lung revealed a diffuse large B-cell-type lymphoma within the bronchiolar wall and alveolar septum. Although this is a rare occurrence, diffuse large B-cell-type non-Hodgkin's lymphoma should be considered in the differential diagnosis of pulmonary diffuse ground-glass opacity in the chest CT scan.

Primary Sjögren's syndrome complicated by sarcoidosis.

A 51-year-old woman suffered from xerophthalmia and xerostomia for 3 years without being medically examined. In July 2006, she was referred to our hospital for the evaluation of chest roentgenogram showing slight pleural effusion in the right lung. A chest CT scan revealed multiple nodules, enlarged mediastinal and hilar lymph nodes, and bilateral slight pleural effusions. A diagnosis of Sjögren's syndrome was made on the basis of the results of sialography, lip biopsy, Schirmer's test, and elevated titer of antibody to SS-A antigen. Histological examination of the specimen from the nodular lesion by video-assisted thoracoscopic biopsy revealed noncaseating epithelioid cell granuloma containing giant cells, which confirmed the diagnosis of sarcoidosis. Although the coexistence of Sjögren's syndrome and sarcoidosis has been reported occasionally, cases with histological evidence of sarcoidosis have been rare. Pulmonary sarcoidosis should be considered in the differential diagnosis of pulmonary multiple nodules in patients with Sjögren's syndrome.

Giant cell tumor of the temporal bone with intratumoral hemorrhage.

We report a case of hemorrhagic giant cell tumor (GCT) of the temporal bone in a 77-year-old woman. The patient suffered from sudden-onset headache and vomiting associated with left temporal hemorrhage. MRI revealed a left temporal extradural mass lesion expanding to the subtemporal fossa, showing strong hypointensity on T(2)-weighted imaging. Subsequent MRI revealed tumor growth with multiple cystic components at 1-month follow up. The tumor was found to be a GCT associated with recent intratumoral hemorrhage and abundant hemosiderin pigmentation. T(2)-weighted MRI of the GCT strongly supported hemosiderin deposition. Secondary formation of cystic components in the GCT can also reflect prior hemorrhage and indicate the progression of shape modification. A literature review revealed that hemosiderin deposition in this rare entity is not as rare as previously thought and that massive intratumoral hemorrhage may occur.

Uterine lipoleiomyoma in an elderly patient: a case report.

BACKGROUND: Uterine lipoleiomyoma is a rare, fat-containing, benign tumor. The clinical features and optimal treatment of this tumor remain unclear. CASE: An 80-year-old woman with an extensive cardiac history was referred for assessment of a huge pelvic mass. Magnetic resonance imaging demonstrated a uterine tumor with inhomogeneously high signal intensity. A fat suppression technique showed heterogeneous suppression of the signal intensity, with areas of increased enhancement after Gd-DTPA administration. Hysteroscopy revealed no abnormal findings in the endometrium. She underwent a simple total abdominal hysterectomy, with a preoperative diagnosis of uterine liposarcoma. Histological examination revealed a uterine lipoleiomyoma. Ten days after surgery, she died due to acute non-occlusive mesenteric ischemia. CONCLUSION: In cases where the radiological appearance of a uterine tumor in an elderly patient is suspicious for liposarcoma, the possibility of a benign lipoleiomyoma should be considered. Careful consideration of the patient's preoperative condition and familiarity with uterine fat-containing tumors are essential for determining an optimal therapeutic approach.

Single-stage operation for a giant haemangiopericytoma following intracranial feeder embolization.

Meningeal haemangiopericytomas (HPC) are malignant intracranial neoplasms that commonly recur and metastasize. Large size at diagnosis, abundant intracranial feeders and the risk of intraoperative bleeding can make them difficult to completely remove in one operation. We report here a rare case of a giant HPC which was treated successfully using a one-stage operation following superselective intracranial feeder occlusion. A 30-year-old man presented with a left middle cranial fossa tumour extending to the left temporal lobe and cerebellar tentorium. Angiography revealed supply from a dilated left posterior temporal artery branching from the posterior cerebral artery. The tumour was totally removed in a single-stage excision after embolization of the intracranial major feeding artery. The present case suggests the usefulness of preoperative embolization for HPC, particularly of intracranial feeders, to achieve total resection safely in a single operation.

Differential expression of RUNX genes in human esophageal squamous cell carcinoma: downregulation of RUNX3 worsens patient prognosis.

BACKGROUND: The RUNX proteins are a family of transcriptional factors that have essential functions during embryogenesis and development, whereas deregulation in expression of RUNXs is often linked to tumor formation. To date, there has been no study describing the precise expression, prognostic impact and methylation status of RUNXs in esophageal squamous cell carcinoma. METHODS: Resected specimens from 61 patients with esophageal SCC were used to identify the expression of RUNX1, RUNX2 and RUNX3 by real-time RT-PCR. Localization of mRNA was done by in situ hybridization, expression of Smad4 was evaluated by immunohistochemistry, and the methylation status of RUNX3 was analyzed by methylation-specific PCR. RESULTS: RUNX3 had a significant impact on patient prognosis with worse survival in the RUNX3-negative group. In early tumors (T1/T2), the prevalence of lymph vessel invasion and the number of metastatic lymph nodes were significantly higher in RUNX3-negative tumors. Furthermore, RUNX3 became a strong prognostic factor only in Smad4-positive tumors. Also, the methylation status of the RUNX3 promoter had a significant correlation with the loss of RUNX3 expression. CONCLUSION: Downregulation of RUNX3 may play a role in disease progression of esophageal SCC, and hypermethylation of the promoter region might be one of the crucial pathways to silence RUNX3 gene.