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Biomedical subjects

T Shimotsuji

Publications and source records attributed to T Shimotsuji.

At least 19 recordsLinked to original sources

Hyperintensity of posterior pituitary on MR T1WI in a boy with central diabetes insipidus caused by missense mutation of neurophysin II gene.

We present a 10-year old boy with central diabetes insipidus (CDI) showing hyperintensity in a normal-sized posterior pituitary on magnetic resonance (MR) T1-weighted image (T1WI). He complained of nocturnal enuresis and polyuria. Daily urine volume increased to 4 to 5 L, and AVP plasma level was very low. Polymerase chain reaction (PCR)-amplified exons of the arginine vasopressin (AVP)-neurophysin (NP) II gene were sequenced. Nucleotide-1884 guanine in Exon 2 was substituted with thymine, which induced a substitution of glycine for valine at amino acid position 65 in the NP II moiety. However, MR imaging showed hyperintensity in the posterior pituitary on T1WI. These results suggest that the MR findings of the posterior pituitary in CDI may vary.

Child↗

[Primary hyperparathyroidism in a child with parathyroid crisis--review of 24 cases reported in Japan].

A 9-year-old boy, who had been admitted to the Pediatric Department for the examination of IgA nephropathy, was transferred to our urological clinic on Aug. 6, 1985, because of parathyroid crisis. Before urological consultation, he had been complaining of bilateral knee and calcaneal pain, anorexia and abdominal pain, which had persisted for several days. Laboratory data indicated serum Ca of 17.6 mg/dl, iP of 2.3 mg/dl and iPTH of 0.77 ng/ml. Roentgenographic examination such as chest, extremities and neck computed tomography showed no abnormal findings. The final diagnosis was parathyroid crisis caused by primary hyperparathyroidism and neck exploration was carried out on August. 10. Left upper parathyroid gland, which was 1 cm in diameter, was surgically removed. The other three glands were normal in size. Histological examination of the resected parathyroid gland revealed chief cell adenoma. In the post-operative course, serum Ca level was soon reduced to the normal range and bone pain disappeared rapidly. During the follow up period of 2.5 years, the patient was clinically free of recurrence. Only 23 cases of primary hyperparathyroidism in children have been reported in the Japanese literature. The clinical characteristics of these specific cases are that primary hyperparathyroidism in children shows a low incidence of renal lesion, but the complicated bone disease is of higher incidence compared with the adult cases. Histopathologically, parathyroid adenoma was frequently observed (14/19), and the other 5 cases were parathyroid hyperplasia.

Adenoma↗

Reduced serum carnosinase activity in hypothyroidism.

Carnosinase hydrolyses carnosine in muscle, and its deficiency is associated with extensive neuromuscular abnormalities. We measured serum carnosinase activity in patients with thyroid dysfunction which often involves neuromuscular systems. In hyperthyroidism, the carnosinase activity was not significantly different from that in normal subjects. In hypothyroidism, however, it was significantly lower than that in normal subjects. The activity examined in five patients with hypothyroidism returned to normal after replacement therapy. In hypothyroidism, the carnosinase activity showed significant correlation with concentration of serum thyroxine and negative correlation with serum creatine kinase activity. This finding may be of practical importance in the differential diagnosis of disorders causing carnosinase deficiency.

Clinical Enzyme Tests↗

[Combination chemotherapy with cis-diamminedichloroplatinum, vinblastine and bleomycin for a rhabdomyosarcoma of the prostate in a child: report of a case].

A case of prostatic rhabdomyosarcoma in a 5-year-old boy is reported. He was brought to our clinic on Apr. 1, 1982 with complaints of pollakisuria and urethral pain. X-ray examinations revealed a huge intrapelvic tumor, and it was histopathologically diagnosed as embryonal rhabdomyosarcoma with a specimen of transrectal needle biopsy. Since the tumor was too huge to resect completely, he was initially treated with combination chemotherapy regimen of vincristine, actinomycin D and cyclophosphamide (VAC therapy), and resulted in failure. Then another combination chemotherapy consisting of cis-diamminedichloroplatinum, vinblastine and bleomycin (PVB therapy) was tried, and the tumor showed reduction in size. On Oct. 15, 1982, total cystectomy with ileal conduit urinary diversion was performed. Histopathologically, degenerative change and partial necrosis of the tumor cell were recognized. After the operation, he was treated with radiation therapy and prophylactic VAC therapy. But six months later, multiple pulmonary metastases occurred and gradually increased in size and number. They did not respond to any other chemotherapy. He died on July 13, 1983. We discussed the chemotherapy for rhabdomyosarcoma, and emphasized that the PVB therapy should be tried on rhabdomyosarcoma as an initial chemotherapy.

Age Factors↗

[Therapeutic effect of cefotaxime in the field of pediatrics].

Cefotaxime (CTX) was administered to 130 children with various bacterial infections of 41 to 400 mg/kg/day for 2 to 21 days. The clinical effect of CTX was very satisfactory in respiratory tract infection, urinary tract infection and meningitis. The overall clinical effect was excellent in 59, good in 39, fair in 17 and failure in 8 with effective rate of 79.7%. During this therapy, side effects were seen in 3 cases, diarrhea in 1 and rash in 2. Abnormal laboratory findings were seen in 5 cases, elevation of GOT in 1, GOT, GPT and A1-P in 1, GOT, GPT and T. Bil. in 1, elevation of BUN, increase of number of basophils and albuminuria in 1 and observation of albuminuria in 1. The above results demonstrate that CTX is a clinically useful antibiotic for the therapy of pediatric infections.

Adolescent↗

Fluorometric assay of human serum carnosinase activity in normal children, adults and patients with myopathy.

A sensitive, accurate method has been established for the assay of serum carnosinase by measuring the fluorescence emitted from the L-histidine liberated on treatment with o-phthaldialdehyde. Using this method the serum values for normal adults, infants and children were measured. The mean value was very low in infants of less than 1 year old but increased with age, being almost the same in children aged 6 years or more as in adults. In adult men, the mean activity was 1.85 mumol/mL/h and in adult women it was 2.07 mumol/mL/h. Low activity was observed in patients with muscular dystrophy.

Adult↗

Serum calcium regulating hormones in the perinatal period.

To clarify perinatal vitamin D metabolism, we measured 25-hydroxyvitamin D (25OHD), 24,25-dihydroxyvitamin D [24,25(OH)2D], 1,25-dihydroxyvitamin D [1,25(OH)2D], calcium (Ca), phosphorus (P), parathyroid hormone (PTH), and human calcitonin (CT) in paired maternal, cord, and infant serum. Cord serum 25OHD was significantly lower than the maternal level, and cord serum 24,25(OH)2D was also significantly below the maternal concentrations. Maternal, cord, and infant serum 1,25(OH)2D, on the other hand, was significantly higher than the normal adult level. The serum PTH was low, but the CT concentration was high in the cord. Cord serum Ca and P levels were significantly higher than maternal. The reason for the elevated circulating 1,25(OH)2D level in the perinatal period is uncertain, and we speculate that the possible factors are gonadal steroids, placental lactogen, prolactin, and CT. In addition, serum 24,25(OH)2D and 1,25(OH)2D concentrations are under some control by the fetus.

Adult↗

Secondary hyperparathyroidism with 1,25-dihydroxyvitamin D deficiency and pseudohypoparathyroidism in childhood: relationship between plasma 1,25-dihydroxyvitamin D and parathyroid hormone levels and urinary cyclic AMP response to exogenous PTH.

In order to clarify the complex interrelationship between serum calcium, 1,25-dihydroxyvitamin D (1,25(OH)2D), and parathyroid hormone (PTH), and the urinary excretion of cyclic AMP (cAMP) in response to exogenous PTH in pseudohypoparathyroidism (PHP) and related diseases, we investigated 3 patients with parathyroid disorders before and after treatment with 1 alpha-hydroxyvitamin D3 (1 alpha-OH-D3). Low plasma 1,25(OH)2D before treatment increased after giving 1 alpha-OH-D3 (0.1 micrograms/kg/day), whereas high plasma PTH measured by the c-terminal assay (C-PTH) decreased in all 3. No response in urinary cAMP was found before or after treatment in 2 patients with PHP type I, despite the fall of plasma C-PTH. However, in one patient with extremely high plasma C-PTH but normal N-PTH (measured by a homologous radioimmunoassay using 1-34 human PTH), urinary cAMP response to exogenous PTH was increased after treatment with 1 alpha-OH-D3. We suggest that he had pseudopseudohypoparathyroidism (PPHP) with Albright's hereditary osteodystrophy and a partial deficiency of renal 1 alpha-hydroxylase. In this patient secondary hyperparathyroidism is thought to be due to 1,25(OH)2D deficiency, and the decreased responsiveness to exogenous PTH before treatment due to excess PTH occupying renal receptors.

Adolescent↗

Plasma active vitamin D concentration in low birthweight infants with rickets and its response to vitamin D treatment.

Impairment of 25-hydroxylation may be a cause of rickets in infants of low birthweight, weighing between 2000 and 2500 g. In addition there may be impairment of 1 alpha-hydroxylation in infants weighing less than 2000 g. Our data show that a supplementary dose of vitamin D2 of at least 500 IU daily is a reasonable regimen for infants who weighed between 2000 and 2500 g at birth. However for infants who weighed less than 2000 g with a gestation of under 38 weeks, administration of 1 alpha-OHD3 may be more effective in preventing rickets.

25-Hydroxyvitamin D 2↗

Effect of dibutyryl adenosine 3',5'-monophosphate administration on plasma concentrations of 1,25-dihydroxyvitamin D in pseudohypoparathyroidism type I.

The effect of dibutyryl cAMP on plasma concentrations of 1,25-dihydroxyvitamin D [1,25-(OH)2D] was studied in two patients with pseudohypoparathyroidism (PHP) type I, five normal adults, and three normal children as controls. In normal adults, plasma 1,25-(OH)2D tended to increase 3 h after the infusion of 2.5 mg/kg dbcAMP and was significantly increased after 6 h. In normal children, plasma 1,25-(OH)2D increased 3 h after the infusion, then gradually decreased; in the patients with PHP type I, it increased greatly from 6.8 and 11.2 pg/ml to 264.6 and 128.2 pg/ml 3 h after the infusion. These results suggest that the renal mechanism for the response to parathyroid hormone is intact distal to the renal adenylate cyclase in patients with PHP type I.

24,25-Dihydroxyvitamin D 3↗

A specific competitive protein binding assay for serum 24,25-dihydroxyvitamin D in normal children and patients with nephrotic syndrome.

A specific competitive protein binding assay for 24,25-dihydroxyvitamin D by Sephadex LH-20 column chromatography, followed by high pressure liquid chromatography with normal rat kidney cytosol as the binding protein, was developed. The mean concentrations of serum 24,25-dihydroxyvitamin D of the cord, in newborn infants and in infants under 12 months of age were 0.90 +/- 0.40 (S.D.) ng/ml, 0.52 +/- 0.21 (S.D.) ng/ml and 1.20 +/- 0.38 (S.D.) ng/ml, respectively. These concentrations were significantly lower than those in children aged 1-15 years (1.96 +/- 0.83 (S.D.) ng/ml). The serum levels in the acute stage of the nephrotic syndrome were significantly reduced, and they increased in remission. These results show that patients wioth nephrotic syndrome have low levels of serum 24,25-dihydroxyvitamin D. This is probably due to its loss in the urine.

24,25-Dihydroxyvitamin D 3↗

Plasma 1,25-dihydroxyvitamin D concentrations in cords, newborns, infants, and children.

Plasma 1,25-dihydroxyvitamin D [1,25-(OH)2-D] was measured in cord serum, newborns, infants, and children. The mean for the values obtained from the six cords was significantly higher than the mean for the older children (6-15 years). The mean for the six newborns (0-1 week) was significantly higher than that for the older children. The mean for the nine infants (1 week-6 months) and the 14 younger children (6 months-6 years) was significantly higher than that for older children. The present study suggests that the perinatal period is associated with a marked increase in 1,25-(OH)2-D.

Adolescent↗

Treatment of hypophosphataemic vitamin D-resistant rickets with massive doses of 1 alpha-hydroxy-vitamin D3 during childhood.

Plasma levels of 1,25 dihydroxy-vitamin D (1,25-(OH)2-D) were low in 3 children with hypophosphataemic vitamin D-resistant rickets (HVDRR) during childhood, but increased after very large doses (0.5 to 2 micrograms/kg per day) of 1 alpha-hydroxy-vitamin D (1 alpha-OH-D3). This treatment has two advantages. Firstly, hypercalcaemia is easily controlled by reducing the dose of 1 alpha-OH-D3 because of its short half-life. Secondly, the administration of 1 alpha-OH-D3 to patients with HVDRR can enhance the tubular reabsorption of phosphate, and this seems desirable in treating HVDRR.

Adolescent↗

Vitamin D metabolism in hypophosphatemic vitamin D-resistant rickets.

Plasma levels of 1,25-(OH)2-D were low in children with hypophosphatemic vitamin D-resistant rickets (HVDRR), but increased after very large doses of 1 alpha-OH-D3. These results suggest that the metabolism of 1,25-(OH)2-D is accelerated in HVDRR. In addition, the lower level of plasma 1,25-(OH)2-D in untreated HVDRR was correlated with the lower level of serum phosphate and renal threshold phosphate concentration (TmP/GFR). The administration of 1 alpha-OH-D3 to the patients with HVDRR could enhance the renal threshold phosphate concentration.

Alkaline Phosphatase↗