Chest wall mesenchymal hamartoma associated with a massive fetal pleural effusion: a case report

We report on an extremely rare chest wall mesenchymal hamartoma associated with a massive fetal pleural effusion. Prenatal ultrasound examination demonstrated a heterogeneous mass in the right thorax associated with a massive pleural effusion and right lung compression at 29 weeks of gestation. The patient underwent pleuroamniotic shunting at 30 weeks and was delivered at 33 weeks by cesarean delivery secondary to fetal distress. After management of the respiratory distress and evaluation of the mass, surgery was performed at day of life 8. Histological examination confirmed the diagnosis of a chest wall mesenchymal hamartoma.     

Beneficial effect of FR183998, a Na+/H+ exchanger inhibitor, on porcine pancreas allotransplantation retrieved from non-heart-beating donors

Activation of Na(+)/H(+) exchanger (NHE) may have an important role in the ischemia/reperfusion injury by producing intracellular calcium overload. Recent studies have shown a beneficial effect of an NHE inhibitor on the ischemia/reperfusion injury in the heart. In this study, we examined the effect of FR183998, a potent NHE inhibitor, in porcine pancreas allotransplantation from non-heart-beating Landrace pig donors (NHBDs). The four experimental groups included: untreated with no preservation (group 1; n = 3), treated with no preservation (group 2; n = 5), untreated with preservation (group 3; n = 6), and treated with preservation (group 4; n = 4). The preservation was made in ice-cold University of Wisconsin (UW) solution for 24 hours. The groups treated received 1 mg/kg FR183998 before donor cardiac arrest and 10 mg in the UW solution flush in situ. Serum blood glucose, insulin, and amylase were measured daily. An intravenous glucose tolerance test (IVGTT) was performed on the postoperative day (POD) 7 when pigs were sacrificed for histological examination. Graft survival rates on that day in groups 1,2,3, and 4 were 3 of 3; 5 of 5; 3 of 6; and 4 of 4, respectively. The mean K values of IVGTT in groups 3 and 4 were 0.78 +/- 0.10 and 1.27 +/- 0.16, respectively, which were significantly different (P < .05). Upon histological examination, pancreatic tissue in group 3 showed more severe edema and necrosis than other groups. FR183998 may be considered beneficial for ischemia/reperfusion injury to pancreatic grafts from NHBDs.     

Influence of the polyol pathway on norepinephrine transporter reduction in diabetic cardiac sympathetic nerves: implications for heterogeneous accumulation of MIBG

Purpose Cardiac scintigraphic studies using ¹²³I-labeled metaiodobenzylguanidine ([¹²³I]MIBG) have demonstrated heterogeneous myocardial accumulation of MIBG in diabetes. The accumulation has been found to correlate with a heterogeneous decrease in the expression of norepinephrine transporter (NET). In diabetic peripheral nerve tissue, polyol pathways are activated and cause nerve dysfunction and degeneration. However, there has been little research on the polyol pathway and cardiac sympathetic nerves. Therefore, to assess the influence of the polyol pathway on cardiac sympathetic nervous function, we investigated the regional accumulation of MIBG and NET protein expression in diabetic model rats treated with aldose reductase inhibitor (ARI) for the blockade of polyol pathways.Methods Rats were given a single intravenous injection of streptozotocin (n=76, STZ-D rats). Starting the day after STZ injection, ARI was administered daily to 42 of the rats for 4 weeks (ARI-D rats). To assess the cardiac sympathetic nervous function, [¹²⁵I]MIBG autoradiographic experiments were carried out. Finally, NET protein expression was assessed with a saturation binding assay.Results The myocardial sorbitol concentration was significantly higher in STZ-D rats than in ARI-D rats. There was no heterogeneous accumulation of MIBG in ARI-D rats. There was a heterogeneous decrease of NET expression in STZ-D rats, but not in ARI-D or control rats.Conclusion The gathered data indicate that the enhanced polyol pathway correlates with the decrease in regional cardiac sympathetic nervous function, and this impairment may lead to the reduction of NET protein in cardiac sympathetic nerves of the diabetic inferior wall.An erratum to this article can be found at     

Rescue revascularization therapy with a stent-in-stent technique for acute intracranial internal carotid artery occlusion

A 45-year-old woman presented with progressive stroke due to occlusion of the left internal carotid artery at the level of the cavernous portion (C3/C4). Revascularization was achieved by stent deployment following percutaneous transluminal angioplasty. Stent thrombosis occurred 7 days after primary stenting; it was successfully treated with the stent-in-stent technique. The patient was able to return to her independent life with no sequelae except for slight hemiparesis. In combination with appropriate antiplatelet treatment, the stent-in-stent technique should be considered as a rescue option in patients with re-occlusion of an intracranial stent.     

Colonic stricture secondary to hemolytic uremic syndrome caused by Escherichia coli O-157

Shiga toxin-producing Escherichia coli (E. coli) is known to be one of the main causes of hemolytic uremic syndrome (HUS). Of the secondary complications of HUS, colonic stricture is relatively rare. We herein report on a Japanese girl that demonstrated sigmoid colon stricture secondary to HUS caused by an infection of E. coli O-157. Severe HUS occurred after the E. coli O-157 infection, so hemodialysis was performed due to renal failure. However, 1 month after recovery from HUS, abdominal symptoms occurred. A contrast study in the colon revealed a sigmoid colon stricture. The stricture was operatively resected. Thereafter, her postoperative course was uneventful. We also review the occurrence of this complication secondary to HUS in the literature.     

Improved Quality of Life and Unchanged Magnetic Resonance Brain Imaging After Living Donor Liver Transplantation for Late-Onset Ornithine Transcarbamylase Deficiency: Report of a Case

We report the case of a 7-year-old girl with ornithine transcarbamylase deficiency whose quality of life (QOL) improved greatly after a living donor liver transplantation (LDLT). Ornithine transcarbamylase deficiency had been diagnosed when she was 2 years old and she finally underwent LDLT, with her father as the donor, when she was 7 years old. The patient had suffered episodes of hyperammonemic encephalopathy ranging from lethargy to coma, treated by hemodialysis twice before LDLT, and her intelligence quotient was borderline for her age. Preoperative magnetic resonance imaging (MRI) showed an atrophic area in the subcortical white matter of the frontal lobe. After LDLT, the patient suffered acute rejection with hyperamylasemia, but not hyperammonemia. Postoperative MRI and quantitative MR spectroscopy showed no changes in the subcortical lesion. She has been followed up carefully for 16 months and has had no further complications or any sign of hyperammonemia.     

Hepatic resection of giant metastatic tumor from clear cell carcinoma of the ovary

All cancer patients, particularly those treated for colorectal cancer, should be monitored for the presence of liver metastases, but liver metastases from ovarian clear cell carcinoma are quite rare. We report a patient subjected to extended left hepatectomy due to a giant metastasis 5 years after surgical treatment for an ovarian neoplasm that was histopathologically diagnosed as clear cell carcinoma. A 58-year-old woman had undergone hysterectomy and bilateral salpingo-oophorectomy due to ovarian cancer (stage Ic). Four years and 8 months after the operation, a computed tomography (CT) scan demonstrated a giant tumor in the left lobe of the liver. The tumor compressed the inferior vena cava (IVC), but it was not clear whether it invaded the vessel. She received chemotherapy for 4 months; however, the tumor did not decrease in size. She was subsequently referred to our institution and was submitted to operation after it was confirmed that there were no distant metastases. After being subjected to an extended left hepatectomy and cholecystectomy, the patient recovered from the surgery without any complications. She has been carefully followed for 17 months and has presented no evidence of recurrence.     

Three cases of pulmonary hamartoma appearing after radical nephrectomy for renal cell carcinoma

We report 3 patients with pulmonary hamartoma, all of whom had undergone nephrectomy for renal cell carcinoma. A lung tumor was detected 2 to 9-months following nephrectomy. Preoperative diagnosis was pulmonary metastasis from renal cell carcinoma and pulmonary tumor resection was performed in each case. There was a 9- to 12-month interval between the detection and resection of the lung tumor. The histological diagnosis of the lung tumor in all three patient was pulmonary hamartoma. Following the resection of the lung tumor, recurrence was not noted in any of the patients.     

Clubbed fingers in a patient with inflammatory gingival hyperplasia

The association of clubbing with miscellaneous diseases and its diagnostic implications are such that its detection should prompt consideration of the underlying etiology. We encountered a 48-year-old woman with clubbed fingers and a cauliflower-like gingival swelling on the hard palate of the upper jaw. There were no conventionally well-known causes for clubbing. Histological examination of gum biopsy specimen revealed a diagnosis of inflammatory gingival hyperplasia. As an etiology of clubbed fingers, gingivitis was suggested, since clubbing was regressed in parallel with remission of the gingivitis after the treatment by extraction of anterior teeth. Possible involvement of an autoimmune process in the pathogenesis was also considered, because of concomitant elevation of serum anti-double strand DNA antibodies. We recommend examination of the oral cavity for search of an inflammatory disease in cases with clubbed fingers, particularly when other common causes are not apparent.