Fetal death following high-dose systemic steroid therapy in a pregnant patient with Vogt-Koyanagi-Harada disease

BACKGROUND: In previous reports, all pregnant women with VKH were successfully treated in the second or third trimester with high-dose systemic corticosterioids without any severe complications. We present a case of fetal death during systemic steroid treatment of a VKH disease patient. RESULTS: A 28-year-old woman in the 30th week of pregnancy was diagnosed with VKH disease. Because of the progression to high bullous retinal detachment in spite of topical corticosteroid therapy, systemic intravenous prednisolone (PSL, 200 mg/day) was begun. On day 18 (PSL, 55 mg/day), sudden intrauterine fetal death occurred. Maternal conditions were normal and there was no apparent abnormality in the umbilical cord, placenta, or fetus; no autopsy was allowed. With gradual tapering of the PSL, the patient's vision was preserved, without recurrence of the VKH disease. CONCLUSIONS: We present a case of fetal death in a 28-year-old pregnant woman being treated with intravenous prednisolone for VKH disease. The cause of the fetal death was not clear. It was reported as an adverse event.     

Cerebellar hemorrhage after supratentorial craniotomy--report of three cases.

We report on three cases of remote cerebellar hemorrhage after supratentorial craniotomy, which had much in common in their computed tomographic, operative, and clinical findings. We speculate that, when the patient is in the supine position, displacement of the cerebellum causes stretching of the superior vermian veins and their tributaries, resulting in tearing of these vessels. Postoperative cerebrospinal fluid overdrainage or massive air reflux into the cranial cavity through the drainage tube may accelerate this process. Meticulous management of the drainage system is necessary to prevent this postoperative complication.     

Lack of cyclic nucleotide regulation of MBCQ-induced relaxation of rat ileal smooth muscle.

The effects of the type V phosphodiesterase (PDE V) inhibitors, MBCQ, zaprinast and dipyridamole, on the relationship between relaxation and cyclic nucleotide content were investigated in rat ileal smooth muscle. Each of MBCQ (0.01-10 microM), zaprinast (0.1-100 microM) and dipyridamole (0.1-100 microM) inhibited carbachol (CCh; 10 microM)-induced contractions in a concentration-dependent manner. When compared with the concentrations of these agents producing 50% relaxation (IC50) of CCh-induced contraction, MBCQ was 14-20 fold more potent than the other agents. The inhibitory potency of these agents against high K+ (65 mM KCl)-induced contractions were similar to that for CCh. MBCQ (1, 10 microM) did not significantly increase the cGMP content above control levels in the presence of CCh (10 microM). Both Zaprinast (1-100 microM) and dipyridamole (1-100 microM) increased the cGMP content of smooth muscle preparations in a concentration-dependent manner. There was a positive correlation between the inhibition of the CCh-induced contraction and the increase in cGMP content elicited by zaprinast and dipyridamole (zaprinast; r=0.72, P<0.05, dipyridamole; r=0.92, P<0.05). However, MBCQ at a concentration which induced a medium-sized relaxation did not significantly increase the cGMP content. Neither MBCQ, zaprinast nor dipyridamole significantly increased the cAMP content of the preparations above control. In summary, it is suggested that the inhibition of CCh-induced contractions by zaprinast and dipyridamole involves increases in cGMP content via inhibition of PDE V. However the inhibition of CCh-induced contraction by MBCQ may not involve cyclic nucleotides in rat ileal smooth muscle.     

Five cases of thrombocytopenia induced by primary human herpesvirus 6 infection

Abstract Five patients suffering from exanthem subitum with thrombocytopenia were confirmed as primary human herpesvirus 6 (HHV-6) infection by serological test. All cases had thrombocytopenia during the acute phase of exanthem subitum. The clinical features of these cases were benign, and all recovered without any specific treatment. Moreover. 4 of the 5 cases showed a mild elevation of hepatic transaminase during the same period, and other viral infections including cytomegalovirus, Epstein-Barr virus, and human herpesvirus 7 were ruled out in these patients. It was speculated that direct inhibition of platelet production by the virus or cytokine induced by the virus-infected cells was the mechanism of the thrombocytopenia induced by primary HHV-6 infection.     

IFR4/MUM1‐positive lymphoma in Waldeyer ring with co‐expression of CD5 and CD10

IRF4/MUM1‐positive lymphoma is a new subgroup of germinal center‐derived B‐cell lymphoma, predominantly involving the Waldeyer ring (WR) in children. CD5 expression is rare in these lymphomas. We report a 7‐year‐old Chinese male with B‐cell lymphoma. Evaluation of his specimen by morphology, immunohistochemistry, and FISH analysis demonstrated IRF4/MUM1‐positive lymphoma with strong and extensive CD5 and CD10 positivity. Despite the lack of t(14;18)(q32;q21) rearrangement, BCL2 protein was expressed. Our report highlights the clinicopathologic features of IFR4/MUM1‐positive lymphoma in WR with co‐expression of CD5 and CD10, and thereby provides insight into this newly recognized disease entity.     

Alterations of retinal pigment epithelium in central serous chorioretinopathy

BACKGROUND: To examine with en face optical coherence tomography (OCT) the alterations of retinal pigment epithelium (RPE) in central serous chorioretinopathy (CSC) and their role in the pathophysiology of CSC. METHODS: We examined retrospectively 30 consecutive eyes of 30 patients with various phases of CSC. All patients underwent fluorescein angiography, indocyanine green angiography, and en face and longitudinal OCT examinations. RESULTS: Of 25 eyes with acute CSC, en face OCT showed RPE abnormalities in 22 eyes (88%) with pigment epithelial detachment (PED) in nine eyes (36%) and a small bulge of RPE in the posterior pole in 15 eyes (60%). Of four eyes with chronic CSC, OCT showed PED in one eye (25%) and a small bulge of RPE in other three eyes (75%). Of 29 eyes with acute or chronic CSC, fluorescein angiography showed characteristic pinpoint leakage in 20 eyes (69%). Of these 20 eyes, a leaking point was located within PEDs in five eyes (25%) and was consistent with the bulge of RPE in nine eyes (45%). With indocyanine green angiography, 28 (97%) eyes showed choroidal vascular hyperpermeability. Ten of the 11 eyes with PED (91%) showed PED within the areas of choroidal vascular hyperpermeability. Sixteen of the 18 eyes with a bulge of RPE (89%) showed the bulge within areas of choroidal vascular hyperpermeability. The one eye in the quiescent CSC exhibited multiple small PEDs within the areas of choroidal vascular hyperpermeability by en face OCT. CONCLUSIONS: En face OCT enables us to detect alterations of RPE in eyes with CSC. Most alterations of RPE were associated with choroidal abnormalities.     

Expression of porcine endogenous retrovirus in peripheral blood leukocytes from ten different breeds

The expression of porcine endogenous retroviruses (PERV) was investigated in primary porcine peripheral blood leukocytes (PBL) of ten different pig breeds. The data suggest that PERV exists in all porcine PBL. A new retroviral element, a foamy‐like pol‐related sequence, was also detected in PBL. Three types of PERV were detected in almost every animal. The breeding of PERV‐free pigs is likely to be difficult. Further studies are required to assess the infectious disease risks associated with xenotransplantation.     

Global and regional abnormalities of left ventricular diastolic filling in hypertrophic cardiomyopathy.

Left ventricular diastolic filling was studied by left ventriculography in 17 patients with hypertrophic cardiomyopathy (HCM). In 8 patients this manifested as a spadelike shape (HCM-S type) on the left ventriculogram, and in 9 patients as a banana shape (HCM-B type). Seven patients were studied as controls. Left ventricular end-diastolic pressure was higher in HCM than in controls. In HCM, peak filling rate was decreased from 1.57 +/- 0.28 of the control to 1.28 +/- 0.20 (p less than 0.05), and the time to peak filling rate was prolonged from 27 +/- 6%DT of the control to 37 +/- 7%DT (p less than 0.01). Serial volume analysis in diastole showed filling fraction was also significantly decreased to 8.2 +/- 4.1 (p less than 0.001) at the 25% diastole time and 29.6 +/- 7.2 (p less than 0.01) at half time, compared with 19.8 +/- 6.3 and 32.8 +/- 5.8 for controls, respectively. In HCM-S type, the rate of distention of left ventricular radial axes at the apical area was lower at early and mid-diastole. In HCM-B type, the rate at the basal area was lower at early and mid-diastole. Results suggest that the left ventricular diastolic filling in HCM was impaired not uniformly but regionally in the hypertrophic area at early and mid-diastole.     

A case of double-chambered right ventricle associated with an interventricular septal aneurysm in an elderly patient

Double-chambered right ventricle is a congential malformation caused by an anomalous muscle band obstructing the right ventricular outflow tract. Most reported cases of this condition have been diagnosed in infants, or adolescents. We encountered a 61-year-old woman with a double-chambered right ventricle, associated with a large interventricular septal aneurysm, which is a rare complication. The right ventricular obstruction was corrected with surgery.