Biomedical subjects
Publications and source records attributed to O Arisaka.
We describe two children who after cardiopulmonary arrest developed hypernatremia at the terminal stage. Urinary antidiuretic hormone concentration was very low, indicating central diabetes insipidus. These cases illustrate the necessity of alertness to the development of central diabetes insipidus in patients with severe hypoxic brain damage.
A very rare occurrence of adult granulosa cell tumor of the ovary (not of the juvenile type) causing precocious pseudopuberty in a six-year-old girl is described. An additional feature of interest was that the tumor appeared entirely cystic. To our knowledge, this condition in such a young premenarchal patient has not been reported previously.
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Our previous study demonstrated that levels of dihomo-gamma-linolenic acid (DGLA) and arachidonic acid in serum total lipids decreased in association with increased plasma levels of prostaglandins E2 (PGE2) and F2 alpha (PGF2 alpha) in patients with insulin-dependent diabetes mellitus. In this study, 11 children with insulin-dependent diabetes mellitus completed a double-blind, placebo-controlled study to assess the effect of dietary supplementation with gamma-linolenic acid (GLA) on serum essential fatty acid and plasma PGE2 and PGF2 alpha levels. GLA was given as the seed oil from the evening primrose (EPO) and all patients received either EPO capsules (containing 45 mg of GLA and 360 mg of linoleic acid) or indistinguishable placebo capsules for 8 months. Initially patients took 2 capsules daily for 4 months then 4 capsules daily for a further 4 months. All patients were assessed at the start of the study, after 4 months and at the end of the study, by measuring serum essential fatty acid and plasma PGE2 and PGF2 alpha levels. After administration of 4 capsules daily the DGLA levels increased and PGE2 levels decreased significantly (p less than 0.01) in the EPO compared with the placebo group. Neither fatty acid nor PGE2 and PGF2 alpha levels were altered by administration of 2 EPO capsules daily. This suggests that the altered essential fatty acid and PG metabolism in diabetes may be reversed by direct GLA supplementation.
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To elucidate the role of antidiuretic hormone (ADH) on water and electrolyte balance in patients with asthmatic attacks, urinary arginine vasopressin (AVP) was assayed in 28 asthmatic patients. In a 3-year-old girl with status asthmaticus who developed a grand mal seizure in association with hyponatremia, urinary AVP levels remained high and fluctuated before convulsion; the cause of the convulsion was considered to be water intoxication due to inappropriate ADH secretion. In 19 of 28 patients with moderately severe asthmatic attacks, increases in urinary AVP levels occurred before treatment (300 +/- 80 pg/ml vs. 40 +/- 24 pg/ml (normal controls), p less than 0.01); elevated AVP levels tended to fall in response to intravenous fluid therapy (appropriate ADH secretion) in 2 of 6 patients, but did not fall (inappropriate ADH secretion) in the remaining patients. It is concluded that inappropriate ADH secretion may occur in asthmatic attacks, and that in such a condition there seems to be a potential risk of water intoxication during fluid therapy, as demonstrated in the present patient.
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We describe a 7-year-old girl with precocious puberty in whom a single large cyst (5 cm) and several small cysts (8-10 mm) in the single remaining ovary were detected by the ultrasound examination. Endocrinological examinations confirmed the diagnosis of central precocious puberty. Pathologic findings after the removal of the cystic lesions revealed that the large cyst was derived from degenerated follicular cysts and the small cysts were identical to follicular cysts: all were considered to have been formed by gonadotropin stimulation. In general, surgical removal of an ovarian follicular cyst in central precocious puberty is inappropriate. However, in this unusual patient who had a degenerated large cyst, surgery seemed to be appropriate because of a previously removed teratoma in the contralateral ovary.
The salivary concentration of 17-hydroxyprogesterone (17-OHP) was determined in 11 patients (aged 6 months to 13 years) with congenital adrenogenital syndrome (AGS) due to C21-hydroxylase deficiency and six healthy controls, in order to assess its value in monitoring treatment of AGS. Salivary 17-OHP was measured by a specific radioimmunoassay after extraction with dichloromethane. Only 100 microliters of saliva was needed for the assay. There was a good correlation (r = 0.93; P less than 0.01) between 17-OHP concentrations in paired saliva and serum samples from the patients and the control subjects. There was a wide scatter in salivary 17-OHP levels in the range between 164 ng/l (1.7 ng/ml in serum) of satisfactorily or overtreated patients and 15,500 ng/l (247 ng/l in serum) of undertreated patients. Levels in normal children were between 200 and 300 ng/l. It is concluded that treatment of congenital AGS (21-hydroxylase deficiency) can be carefully monitored in children with the noninvasive, frequently repeatable measurement of salivary 17-OHP.
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A 3-year-old girl with status asthmaticus developed a grand mal seizure in association with hyponatraemia after 16 h of fluid therapy. Urinary arginine vasopressin (AVP) was elevated during the attack and rose strikingly before the onset of the convulsion. In 13 of 17 other patients with moderately severe asthmatic attacks, increases in urinary AVP levels occurred before the initiation of treatment. Dilutional hyponatraemia (water intoxication) must be prevented in patients with severe asthmatic attacks in whom diuretic capacity is impaired.
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A 7-year-old boy developed acute, severe hypercalcaemia following the partial excision of a cerebellar medulloblastoma. The serum calcium level was extremely high (19.8 mg/100 ml), but a skeletal survey revealed no apparent bone metastatic lesions; such lesions were only detected by X-ray 3 weeks after the onset of hypercalcaemia. Hypercalcaemia was promptly resolved by intravenous mithramycin administration, before which the serum parathyroid hormone level, 1,25-(OH)2-vitamin D level and the nephrogenous cyclic AMP level were low. However, the relation between serum calcium levels and urinary calcium excretions indicated that renal calcium reabsorption was increased in association with hypercalcaemia, suggesting that a parathyroid hormone-like effect was operative on the renal tubules. It is possible that a combination of increased bone resorption by metastatic tumour cells and renal tubular handling of calcium presumably mediated by tumour-produced humoral factors was responsible for the acute development of severe hypercalcaemia in this patient with medulloblastoma.
Two patients with hypothyroidism had detectable serum levels of thyrotropin binding inhibitor immunoglobulin (TBII). Patient 1 was a newborn infant who had transient neonatal hypothyroidism due to transfer of TBII from the mother with nongoitrous autoimmune thyroiditis. Patient 2 was an 8-year-old girl with Down's syndrome who presented with signs of myxedema and central precocious puberty. She had no goiter, and the recognition of thyroid disease was delayed; the histological diagnosis of chronic lymphocytic thyroiditis was established by aspiration biopsy, and TBII had strong thyroid adenyl cyclase-inhibiting activity in vitro. It appears that TBII may be pathogenetically important for occurrence of neonatal hypothyroidism and nongoitrous autoimmune thyroiditis without goiter.