Asymptomatic giant cell hepatitis: a subtype of post-infantile giant cell hepatitis?

Giant cell hepatitis in adults is considered a rapidly progressive and life-threatening disease, but there are few descriptions of a prolonged disease course. A 36-year-old Japanese man was referred to our hospital for further evaluation of abnormal liver function test results. Although asymptomatic, he had undergone follow-up for 9 years with these abnormalities. Because the cause of liver injury was not identified despite extensive noninvasive examinations, the patient underwent needle biopsy. He was finally diagnosed with post-infantile giant cell hepatitis (PIGCH) based on the presence of small numbers of giant multinucleated hepatocytes scattered primarily around the portal area. Necroinflammatory changes were very mild in the portal tracts and hepatic parenchyma. According to the histological findings as well as the accepted international diagnostic scoring system for autoimmune hepatitis (AIH), which is closely related to PIGCH, AIH was unlikely, although antinuclear antibody was positive at a titer of 1:160. The present case may describe an unknown subtype of PIGCH, characterized by insidious disease onset and progression with concurrent, mildly active underlying hepatitis, which is in contrast with the well-documented aggressive nature of PIGCH.

Progressive atrophy in a deformed liver as a contributor to sigmoid volvulus.

The effect of a prior defect on secondary liver atrophy is unknown. We describe a case of sigmoid volvulus that was facilitated by progressive atrophy in a deformed liver. A 75-year-old man with abdominal pain and fullness was referred to our hospital. Computed tomography (CT) revealed reduced left hepatic lobe volume and a whirl sign, characteristic of sigmoid volvulus. The sigmoid volvulus was successfully detorted with endoscopy. Retrospective evaluation of liver morphology on CT and magnetic resonance imaging showed that the portal vein at the liver hilum was denuded due to a parenchymal defect of the medial segment, with compression by the crossing artery. As pulse Doppler ultrasonography demonstrated reduced portal blood flow in the region where liver atrophy developed, compression of the denuded portal vein presumably facilitated secondary atrophy and contributed to sigmoid volvulus. The present case shows that a deformed liver itself can be a cause of secondary atrophy. Therefore, continued monitoring of liver morphology and evaluation of portal blood flow to predict liver atrophy may be required, when an individual with a partial liver defect is encountered.

Acute peri-myocarditis with an unusual initial manifestation of gallbladder edema and a profound eosinophilic surge during convalescence.

A 29-year-old man with fever and right upper quadrant pain was referred to our hospital. Ultrasonography revealed intense gallbladder edema and pericardial effusion. Despite no symptoms and signs related to heart failure, the patient was clinically suspected of cardiac dysfunction related to acute peri-myocarditis based on his symptoms of preceding fever and headache, pericardial effusion, positive troponin T value, elevated B-type natriuretic peptide level, and sequential changes on electrocardiography. With a profound eosinophilic surge (8,022/µL) during convalescence, acute peri-myocarditis and gallbladder edema resolved spontaneously. This case instructively shows that acute peri-myocarditis initially manifests with abdominal symptoms, mimicking acute acalculous cholecystitis. In addition, an extensive review of acute myocarditis cases with peripheral eosinophilia suggests that there is a subgroup characterized by a predilection for young and middle-aged men, concurrence of pericardial effusion, transient eosinophilic surge during convalescence, and favorable outcome.

Spontaneous Celiac and Splenic Artery Dissection.

Dissection of the splanchnic artery unrelated to an aortic lesion is extremely rare. We describe a patient with dissection of the celiac and splenic arteries causing splenic circulatory impairment. A 55-year-old Japanese man was referred to our hospital for left back pain that suddenly occurred 3 days previously and spread to the left flank. He had complicated sleep apnea syndrome well controlled with continuous positive airway pressure, and had been prophylactically taking aspirin (100 mg/day) because of asymptomatic cerebral lacunar infarcts. Contrast-enhanced computed tomography (CT) in the arterial phase revealed dissection from the celiac root extending to the entire splenic artery, the caliber of which was irregularly narrowed, causing malperfusion in the spleen. Because of hemodynamic stability and lack of impending sequelae, the patient was carefully observed with rest, strict blood pressure control, and aspirin administration. One month later, CT revealed restoration of the caliber of the dissected arteries and regression of the organizing false lumen, which confirmed the patient's recovery. Despite the extreme rarity or nonspecific symptoms, splanchnic artery dissection should be considered a potentially life-threatening emergency. This case supports the possible benefit of starting antithrombotic treatment early to prevent thrombotic sequelae such as organ infarction and aneurysmal formation.

Follicular lymphoma-related colitis resembling ulcerative colitis.

Among the various manifestations of colonic involvement in malignant lymphomas, an ulcerative colitis-like appearance is rare. Herein, we describe a case in which extranodal colonic involvement resembled ulcerative colitis in a patient with advanced follicular lymphoma. A 59-year-old Japanese man with diarrhea and body weight loss was referred to our hospital. Computed tomography (CT) revealed systemic lymphadenopathy, splenomegaly, and thickening of the sigmoid colon and rectum walls. 18F-fluorodeoxy-glucose positron emission tomography (18F-FDG-PET/CT) revealed intense tubular 18F-FDG accumulation extending from the rectum to the colon at the hepatic flexure and much weaker accumulation in the systemic lymph nodes, bone marrow, and spleen. The isotope-enriched areas had an ulcerative colitis-like appearance as shown via colonoscopy. The patient was ultimately diagnosed with follicular lymphoma (stage IV A, grade 1) based on a pathological examination of the neck lymph nodes, iliac bone marrow, and colon. After six courses of chemotherapy (R-CHOP), 18F-FDG-PET/CT confirmed complete remission of the lymphoma including the colonic lesion. This is presumably the first case of ulcerative-like colitis caused by a follicular lymphoma. As a novel approach, the lymphoma-related colitis was detected by comparing the pathology results and the 18F-FDG-PET/CT results.

Achilles tendinitis as a rare extraintestinal manifestation of ulcerative colitis.

Patients with inflammatory bowel disease often have extraintestinal manifestations (EIMs) involving almost all organ systems, but little has been reported on Achilles tendinitis. Herein, we present a unique case of Achilles tendinitis, which manifested shortly after initiation of mesalazine therapy for ulcerative colitis. A 26-year-old Japanese woman with bloody diarrhea and abdominal cramps lasting for 7 days was referred to our hospital. The Lichtiger clinical activity index (CAI) score was 9 at the first visit. Based on the clinical symptoms and examination results, she was diagnosed with ulcerative pancolitis in the active phase, and treatment with mesalazine (2.4 g/day) and probiotics was initiated. Her symptoms resolved within 7 days of treatment (CAI 3). However, she then developed bilateral Achilles tendinitis without any apparent cause. The Achilles tendinitis subsided with conservative management within 2 weeks, despite continuation of mesalazine therapy. This case instructively suggests that Achilles tendinitis should be noted as an EIM of ulcerative colitis.

Possible Contribution of a Diverticulum to the Development and Rupture of Colonic Lymphangioma.

A 55-year-old Japanese man with a history of diverticulitis underwent colonoscopy for careful evaluation of progressive anemia. A 5-mm depressed lesion oozing spontaneously was observed at the hepatic flexure. On suspicion of depressed-type of cancer, right-sided hemicolectomy was performed. Histopathological examination indicated a collapsed lymphangioma exactly over a diverticulum, which had previously been complicated diverticulitis. The colonic mucosa and lymphangioma prolapsed beyond the subserosal layer via the muscularis propria defect, resulting in a depressed lesion and mucosal laceration with hemorrhage. This case suggests the contribution of a colonic diverticulum to the development and rupture of lymphangioma, which needed to be distinguished from depressed-type colon cancer.

Endoscopic assessment of reflux esophagitis concurrent with hiatal hernia in male Japanese patients with obstructive sleep apnea.

OBJECTIVE: The pathogenetic relationship underlying the high prevalence of gastroesophageal reflux disease (GERD) in patients with obstructive sleep apnea (OSA) remains unclear. In addition, GERD has not been adequately assessed by endoscopy in patients with OSA. The purpose of this study was to use endoscopy to investigate potential interactions among reflux esophagitis, hiatal hernia (HH) and OSA. MATERIAL AND METHODS: A total of 243 consecutive male Japanese participants who underwent both overnight ambulatory polygraphic monitoring and esophagogastroduodenoscopy were retrospectively evaluated in a cross-sectional study. The prevalence and severity of HH and reflux esophagitis were assessed according to the Los Angeles classification and the Makuuchi classification, respectively. Associations among reflux esophagitis, HH and OSA were examined by univariate and multivariate analyses. RESULTS: OSA was diagnosed in 98 individuals (40.3%). Endoscopy-confirmed esophagitis (p = 0.027) and HH (p < 0.001) were significantly more prevalent among patients with OSA. Multivariate regression model analysis adjusted for age, body mass index, visceral obesity represented by waist circumference, presence of OSA, concurrence of OSA and HH, smoking, and alcohol consumption yielded OSA as the only variable significantly associated with HH (odds ratio [OR], 2.60; 95% confidence interval [CI], 1.35-4.99; p = 0.004), while concurrence of OSA and HH was related to reflux esophagitis (OR, 3.59; CI, 1.87-6.92; p < 0.001). CONCLUSIONS: OSA was associated with HH and concurrent OSA and HH with reflux esophagitis in male Japanese patients with OSA. Our results support the hypothesis that complicating HH may link reflux esophagitis to OSA.

Norovirus gastroenteritis accompanied by marked elevation of transaminases.

A 56-year-old woman was admitted because of frequent watery diarrhea, vomiting, and abdominal cramps. An examination of the stool for norovirus antigen was positive, and a blood examination revealed a marked elevation of liver enzymes. Liver dysfunction, as well as symptoms related to gastroenteritis, was ameliorated solely by supportive treatment. Although liver injury concurrent with norovirus gastroenteritis is rarely documented and its pathogenesis remains unknown, clinicians should consider liver injury as one of the possible extra-intestinal manifestations of norovirus gastroenteritis.

Intravascular gas in multiple organs detected by postmortem computed tomography: effect of prolonged cardiopulmonary resuscitation on organ damage in patients with cardiopulmonary arrest.

A 76-year-old woman was found in cardiopulmonary arrest with her head submerged in water in a bathtub. Despite cardiopulmonary resuscitation (CPR) for over 1 h by professional emergency technicians and medical doctors, the patient died. Postmortem computed tomography revealed not only pulmonary edema associated with drowning but also the presence of intravascular gas in the pulmonary artery, liver, kidneys, heart (right ventricle), and brain. It was speculated that intravascular gas was generated and spread to multiple organs during CPR procedures via the alimentary tract and lungs, which had been damaged by ischemia after cardiopulmonary arrest. Prolonged CPR procedures may involve the risk of additional organ damage and systemic air emboli.

Norovirus gastroenteritis accompanied by ischemic colitis: a case report.

A 57-year-old woman was admitted to our hospital because of frequent bloody diarrhea, vomiting, and abdominal cramps. While the patient was diagnosed with norovirus gastroenteritis based on the presence by chance of positive norovirus antigen in the stool samples, endoscopic as well as pathological examinations demonstrated left-sided ischemic colitis. The patient soon recovered solely by supportive treatment. Although it is believed that patients with viral gastroenteritis do not develop bloody diarrhea, the present case suggested that conditions of viral gastroenteritis, such as intensified peristalsis, elevation of the intraluminal gut pressure, hypovolemia, and hemoconcentration might trigger ischemic colitis resulting in bloody diarrhea. Physicians should not exclude the diagnosis of viral gastroenteritis simply by depending on the presence of bloody diarrhea. Further studies to clarify the incidence of ischemic colitis in patients with viral gastroenteritis are indispensable.

[Diffuse large B-cell lymphoma initially manifested by massive ascites and a small gastric lesion, clinically mimicking primary effusion lymphoma (PEL) in the abdominal cavity: a case report and review of the literature on Japanese PEL patients].

A 50-year-old man was admitted because of increasing massive ascites. While lymphoma cells (diffuse large B-cell lymphoma: DLBCL) were detected in the ascites, pleural effusion, cerebrospinal fluid and bone marrow, no tumor masses other than a submucosal lymphoma lesion in the stomach only 1 cm in diameter were identified. The patient was treated with chemotherapy including rituximab (R-CHOP-ESHAP) and injection of methotrexate and dexamethasone into the medullary cavity as well as radiation to the whole brain, and achieved complete remission 4 months later. The present case suggests that DLBCL can initially manifest as a form of effusion lymphoma with minimum solid tumor component. The distinctive clinical features of Japanese patients with primary effusion lymphoma are also reviewed.

Metastatic colon cancer mimicking Crohn's disease.

A 66-year-old Japanese woman presented with intestinal obstruction and right-sided hydronephrosis. Although upper gastrointestinal endoscopy demonstrated signet-ring cell gastric carcinoma, colonoscopy and barium enema study yielded findings mimicking Crohn's disease in the colon, that is, skipping longitudinal ulcer scarlike strictures, cobblestone appearance, segmental stricture, and pseudosacculations. After total gastrectomy and right-sided hemicolectomy, the final diagnosis of gastric cancer extensively involving the colon, and not of Crohn's disease complicating gastric cancer, was established. Pathologic examination showed that anaplastic cancer with exuberant desmoplastic reaction and infiltration along the mesenteric border principally accounted for the morphological similarities noted between Crohn's disease and metastatic colon cancer in this case. The findings in the present case, together with a review of the literature, suggest that metastatic colon cancer should be considered when Crohn-like colonic findings are encountered, not only in individuals with concurrent cancer in other sites but also in those with distant history of cancer.

A variant of thyrotoxicosis associated with chronic thyroiditis characterized by prolonged fever, absence of anti-thyroidal antibodies, and favorable response to naproxen.

An unusual form of thyrotoxicosis due to chronic thyroiditis is described. A 78-year-old debilitated woman was admitted because of fever to 38 degrees C persisting for the previous 16 months, for which the antipyretic effect of diclofenac sodium and pranoprofen had been insufficient or transient. Intense accumulation of gallium-67 citrate in the thyroid gland provided an initial clue to the diagnosis of masked thyrotoxicosis as a cause of the fever, and naproxen (300 mg/d) eliminated the fever and flaring of thyrotoxicosis. Despite the absence of autoantibodies related to chronic thyroiditis, needle biopsy revealed destructive thyroiditis due to chronic lymphocytic thyroiditis. This case suggests a previously unrecognized variant of thyrotoxicosis due to chronic thyroiditis, that is, neither painless thyroiditis nor acute exacerbation of Hashimoto thyroiditis, and instead characterized by prolonged fever, nontender thyroid, absence of antibodies associated with autoimmune thyroiditis, and excellent response to naproxen.

Serum insulin levels and the prevalence of adenomatous and hyperplastic polyps in the proximal colon.

BACKGROUND & AIMS: Obesity and diabetes mellitus are associated with an increased incidence of proximal colon cancer. Colonic adenoma that has been reported to be associated with elevated serum insulin levels and subsets of hyperplastic polyps might serve as a precursor of colon cancer. In this study, we sought to determine segment-specific associations between serum insulin levels and the prevalence of adenoma and hyperplastic polyps in the proximal and distal colon. METHODS: We studied 343 consecutive patients who underwent colonoscopy in our hospital. All medical information, including fasting serum insulin, was obtained at colonoscopy. We performed multinomial logistic regression models by using the outcome categories of none (reference), proximal-only, distal-only, and both-segment lesions for the presence of adenoma/hyperplastic polyp with serum insulin, age, gender, lifestyle characteristics, and the presence of other types of lesions as predictors. Odds ratios (ORs) and 95% confidence intervals (CIs) are provided for a 5-muU/mL increase in serum insulin. RESULTS: Overall, serum insulin levels were significantly associated with adenoma (OR, 1.5; 95% CI, 1.1-2.0; P = .005) and borderline associated with hyperplastic polyps (OR, 1.3; 95% CI, 1.0-1.7; P = .075). In multinomial logistic regression models, elevated serum insulin levels were significantly associated with proximal-only adenoma (OR, 1.8; 95% CI, 1.2-2.5; P = .002), both-side hyperplastic polyp (OR, 1.7; 95% CI, 1.1-2.5; P = .015), and proximal-only hyperplastic polyp (OR, 1.5; 95% CI, 1.0-2.1; P = .048) and borderline associated with distal-only adenoma (OR, 1.5; 95% CI, 1.0-2.1; P =.059) but not with distal-only hyperplastic polyp. CONCLUSIONS: Serum insulin levels directly correlate with the presence of adenoma and hyperplastic polyps in the proximal colon and might also less strongly correlate with the presence of distal adenoma.