A case of primary hyperparathyroidism with severe bone and renal changes

We report a 79-year-old Japanese woman who had primary hyperparathyroidism (HPT) with end-stage renal disease and severe bone changes. In 2004, she began to experience pain in her shoulders and knees, as well as muscle weakness and anorexia. She already had renal failure with a serum Cr of 4.7 mg/dl, while serum calcium was 9.6 mg/dl, PTH was 2,710 pg/ml, and serum alkaline phosphatase was 923 mU/ml. Multiple fractures of the pelvic bones and lumbar spine, osteoporosis, and subperiosteal bone resorption were detected. Although hemodialysis (HD) was started in February 2005, her symptoms became more severe. Total parathyroidectomy (PTX) and right iliac crest bone biopsy were performed. Histomorphometric analysis of the cancellous bone indicated a diagnosis of osteitis fibrosa, but a reduction of cortical bone and near absence of cancellous bone were also apparent. This showed that bone resorption by osteoclasts was predominant over bone formation by osteoblasts. Soon after PTX, her pain subsided completely. We conclude that primary HPT should be detected and treated early enough to avoid renal damage, since renal dysfunction markedly accelerates bone changes in patients with primary HPT.     

Synovial osteochondromatosis of the hip treated through a surgical dislocation

We report a case of osteochondromatosis in the hip joint treated through a surgical dislocation and describe the use and safety of the procedure confirmed by postoperative magnetic resonance imaging. A 44-year-old man had right hip pain with moderate limitation of the range of motion. Plain radiographs and computed tomography showed calcified loose bodies in the hip with mild joint degeneration. Magnetic resonance imaging showed remarkable expansion of the capsule containing joint fluid and loose bodies below the femoral head. Complete removal of loose bodies and synovectomy were performed through a surgical dislocation. Three months after the operation, the patient had fully recovered and returned to his job. Magnetic resonance imaging performed 5 months after surgery revealed full removal of the loose bodies and no recurrence of the disease including synovial proliferation. It also showed no evidence of avascular necrosis, meaning that the procedure had been performed safely.     

CXCL12 and CCL20 play a significant role in mucosal T-lymphocyte adherence to intestinal microvessels in mice

OBJECTIVE: Although it is known that the chemokines CXCL12 and CCL20 are expressed in the intestine, their contribution to lymphocyte homing has not been investigated in detail. The authors investigated whether the CXCL12-CXCR4 and CCL20-CCR6 systems are involved in T lymphocyte-endothelial interaction in microvessels of the small and large intestines. METHODS: Labeled lamina proprial lymphocytes (LPLs) were administered to mice, and their adhesion to microvessels of normal and TNF-alpha -induced inflamed intestinal mucosa was observed under an intravital microscope. Antibodies against CXCL12, CCL-20, or CCL-25 were administered prior to lymphocyte administration, and in some experiments CXCR4 or CCR6 on LPLs was desensitized with an excess amount of chemokine. RESULTS: LPLs adhered to microvessels of the ileum and colon, and TNF-alpha induced a significant accumulation at both sites. Blocking of the CXCL12-CXCR4 system significantly inhibited the LPL adhesion in the ileum and colon under both normal and TNF-alpha -treated conditions. However, blocking of the CCL20-CCR6 system significantly attenuated LPL adhesion only under a TNF-alpha -treated condition. There was an additive inhibitory effect on LPL adherence by CXCL12 and CCL20 blocking in TNF-alpha -induced inflamed intestines. There was also an additive function of the CCL25-CCR9 system in LPL accumulation in the small intestine. CONCLUSION: Several chemokine systems may play significant roles cooperatively in vivo in LPL adherence to microvessels of intestinal mucosa.     

Application of the recent American practice resources for risk stratification system for patients presenting to a Japanese emergency department because of syncope

BACKGROUND: The American College of Physicians (ACP) and the American College of Emergency Physicians (ACEP) recently published practice guidelines and recommendations for evaluation of patients with syncope based on historical, physical, and ECG findings. The objective of the present study was to determine if risk stratification using these practice resources is valid in a series of Japanese patients. METHODS AND RESULTS: A total of 912 consecutive patients brought to our emergency department between 1988 and 1997 because of syncope were identified. Follow-up information about mortality was obtained for 707 patients by means of mailed questionnaires and from medical records, and the mortality data were analyzed by the actuarial life-table method. A total of 187 patients who fulfilled the admission criteria according to the ACP guidelines were found to have higher overall and cardiac mortality than the other 520 patients (P < 0.0001), and 153 patients who fulfilled the admission criteria according to the ACEP recommendations also had higher overall and cardiac mortality than the other 554 patients (P < 0.0001). CONCLUSIONS: The recent American practice recommendations can be used for risk stratification of syncope patients in Japan. Historical, physical, and ECG findings available on presentation can be used to stratify the risk of mortality in patients brought to Japanese emergency departments because of syncope.     

A case of eosinophilic cholangitis： Imaging findings of contrast-enhanced ultrasonography, cholangioscopy, and intraductal ultrasonography

A 38-year-old woman was referred to our institution due to epigastralgia. She presented with obstructive jaundice and eosinophilia. Endoscopic retrograde cholangiopancreatography showed diffuse narrowing from the distal common bile duct to the bifurcation of the hepatic ducts. An endoscopic plastic biliary stent was inserted; the specimen obtained from the common bile duct wall revealed dense infiltration by eosinophils. Treatment was started with prednisolone 60 mg daily. The patient＇s biliary stenosis and eosinophilia gradually improved. Eosinophilic infiltration in the lungs or stomach is relatively common, but it is rare in the common bile duct. Most of the reported cases of eosinophilic cholangitis presented with eosinophilia; our patient＇s eosinophil count was over 1000/mm^3. Since our patient had allergies to pollen and house dust, a relationship between the allergies and the eosinophilic cholangitis was suspected, but no cause was identified.     

Changes in the function of the inhibitory neurotransmitter system in the rat brain following subchronic inhalation exposure to 1-bromopropane

1-Bromopropane (1-BP) has been widely used as a cleaning agent and a solvent in industries, but the central neurotoxicity of 1-BP remains to be clarified. In the present study, we investigated the effects of subchronic inhalation exposure to 1-BP vapor on the function of the inhibitory neurotransmitter system mediated by gamma-aminobutyric acid (GABA) in the rat brain. Male Wistar rats were exposed to 1-BP vapor for 12 weeks (6h/day, 5 days/week) at a concentration of 400 ppm, and, in order to investigate the expression and function of brain GABA type A (GABAA) receptors, total/messenger RNA was prepared from the neocortex, hippocampus, and cerebellum of the control and 1-BP-exposed rats. Moreover, hippocampal slices were prepared, and the population spike (PS) amplitude and the slope of the field excitatory postsynaptic potential (fEPSP) were investigated in the paired-pulse configuration of the extracellular recording technique. Using the Xenopus oocyte expression system, we compared GABA concentration-response curves obtained from oocytes injected with brain subregional mRNAs of control and 1-BP exposed rats, and observed no significant differences in apparent GABA affinity. On the other hand, paired-pulse inhibition of PS amplitude was significantly decreased in the hippocampal dentate gyrus (DG) by exposure to 1-BP, without any effect on the paired-pulse ratio of the fEPSP slopes, suggesting neuronal disinhibition in the DG. Moreover, RT-PCR analysis indicated decreased levels of GABAA receptor beta3 and delta subunit mRNAs in the hippocampus of 1-BP-exposed rats. These results demonstrate that subchronic inhalation exposure to 1-BP vapor reduces the function of the hippocampal GABAergic system, which could be due to changes in the expression and function of GABAA receptors, especially the delta subunit-containing GABAA receptors.