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Biomedical subjects

H Isotani

Publications and source records attributed to H Isotani.

28 records · Page 2Linked to original sources
PubMed1996

Diabetic ketoacidosis associated with the pheochromocytoma of youth.

We report a case of diabetic ketoacidosis (DKA) associated with pheochromocytoma in a college student of 22 years of age who was admitted to hospital because of hyperglycemia and hypertension with palpitations and subsequently was found to have an adrenalin and noradrenalin secreting pheochromocytoma. A diagnosis was made and the patient's impaired glucose tolerance and hypertension improved by tumor excision. This is the first reported case of a noradrenalin-predominant pheochromocytoma with associated DKA. Although DKA has been thought not to occur with pheochromocytoma, the possible cause of DKA associated with pheochromocytoma is now described. It is important to point out, as a clinical manager, that severe hypertension is a very unusual concomitant of DKA in young people and is the main indication for further examination.

Adrenal Gland Neoplasms↗
PubMed1996

Hypoparathyroidism and insulin-dependent diabetes mellitus in a patient with Kearns-Sayre syndrome harbouring a mitochondrial DNA deletion.

We report a 17-year-old girl with short stature, external ophthalmoplegia, atypical retinal pigmentary degeneration, sensorineural hearing loss, and cardiac conduction defect (Kearns-Sayre syndrome). A large-scale deletion (6741 base pairs) in mitochondrial DNA was found in her muscle specimen. She also had insulin-dependent diabetes mellitus (IDDM). On admission, her plasma glucose level was elevated at 31.0mmol/l with mild ketoacidosis, and haemoglobinA1c elevated at 16.5%. After improvement of diabetic ketoacidosis, she was placed on insulin 24-30 units/day despite her small body weight of 25 kg. There was reduced excretion of urinary C-peptide at 3.97 nmol/day. In addition, she had idiopathic hypoparathyroidism with a serum calcium level of 2.15 mmol/l, phosphate 1.7 mmol/l, and intact PTH below 10 ng/l. Human leucocyte associated antigen typing showed A24, A26; B54, B61; CW1, CW3; DR8, DR14; DQ1 and DQ3, suggesting that the presence of HLA-A24 and CW3 antigen contributed to the association of IDDM and hypoparathyroidism, similar to Japanese patients with polyglandular autoimmune syndrome, complicated by hypoparathyroidism and IDDM. We suggest that a genetic linkage, as well as mitochondrial dysfunction, may be responsible for the association of the two disease states. This is an extremely rare case of Kearns-Sayre syndrome, presenting in association with IDDM and idiopathic hypoparathyroidism.

Adolescent↗
PubMed1995

[A case of polyglandular autoimmune syndrome (PGA) type 1 complicated with slowly progressive IDDM].

A case of an 18-year-old female with polyglandular autoimmune syndrome (PGA) type 1 complicated by slowly progressive IDDM was described. She had epilepsy at the age of 5, and mucocutaneous candidiasis and hypoparathyroidism at 7 years. At the age of 18, the patient noticed thirst and body weight loss. On admission, she had uneven teeth and chronic mucocutaneous candidiasis. Plasma blood glucose was 312 mg/dl without ketosis, hemoglobin Alc 9.1%, serum calcium 3.5 mEq/l, serum phosphorus 6.0 mg/dl. A CT scan of her brain revealed calcification in the bilateral basal ganglia. Serum intact PTH was less than 10 pg/ml. Ellsworth-Howard's test showed hyperresponsiveness in the secretion of urinary phosphorus and cyclic-AMP. Other endocrinological studies showed no abnormality except for mild hyporesponsiveness in the secretion of urinary C-peptide (39.6 mu g/day). After admission, she was initially treated with diet alone with positive islet cell antibody (ICA). Three months later she was treated with low dose insulin, and ICA became negative. Then 5 months later it became positive again. Sixteen months later she had IDDM with positive ICA and without the secretion of urinary C-peptide. On the basis of these results, we diagnosed this case as PGA type 1 with the manifestations of hypoparathyroidism, chronic mucocutaneous candidiasis and slowly progressive IDDM. This is the second case report in Japan about PGA type 1. Furthermore, this case demonstrates for the first time in Japan that slowly progressive IDDM is complicated by PGA type 1. The patient had this HLA typing: A 24(9), BW52(5), BW60(40), CW3, DR2, DRW12, DQW7. More investigation is necessary to clarify the mechanism of PGA type 1.

Adolescent↗
PubMed1995

Oval pupil in patients with diabetes mellitus: examination by measurement of the dark-adapted pupillary area and pupillary light reflex.

Determination of the dark-adapted pupillary area (DAPA) by infrared photography revealed that some diabetic patients show corectopia (oval pupil) in addition to the small DAPA as pupillary abnormalities. The prevalence and clinical details of oval pupil were compared between diabetic patients and healthy subjects. Pupillary light reflexes were also analyzed with an infrared videopupillography in some of the diabetic patients. The healthy subjects were examined for the influence of age on the ratio of the pupillary diameter of the major and minor axes. The ratio was 1.04 +/- 0.02 (mean +/- S.D.) in the healthy subjects, and cases in which the ratio was +2 S.D. or higher than the mean ratio were defined as oval pupil. Oval pupil was observed in 21 (24%) of 86 diabetic patients, and was correlated with heart rate variation (P < 0.05) and DAPA (P < 0.01), which represent an autonomic dysfunction. Quantitative analysis of pupillary light reflexes with an infrared videopupillography revealed that the dark-adapted pupillary area before photic stimulation (P < 0.01) and the maximum %-velocity of constriction (P < 0.05) were significantly less in the oval pupil group than in the round pupil group. Cranial magnetic resonance imaging revealed no abnormalities in the oval pupil group. From these results, a peripheral autonomic disorder was implicated in the etiology of oval pupils in diabetic patients.

Adult↗
PubMed1991

Insulin-dependent diabetes mellitus in which glycemic control was improved during pregnancy but deteriorated after delivery with the occurrence of postpartum thyrotoxicosis: a case report.

We report a patient, a twin, with diabetes mellitus whose hyperglycemic state fluctuated during the course of the pregnancy and the subsequent delivery. She was diagnosed as having slowly progressive IDDM because of her clinical course and the findings of serum positive ICA/CF, positive HLA-DR4 and disconcordance of diabetes mellitus with her identical twin. Insulin therapy was not initially needed in the first two years because the endogenous insulin secretion was not completely reduced. After two years of insulin therapy the patient became pregnant. Her glycemic control was remarkably improved without changes in dietary intake and insulin dosage. After delivery glycemic control deteriorated after delivery with the occurrence of postpartum thyroiditis. Urinary excretion of CPR was increased during pregnancy but decreased after delivery. ICA/CF in serum were persistently detected in the whole observation period. It seems that the improved glycemic control during pregnancy was caused by the reduction in the autoimmune reaction and the deterioration in glycemic control during the postpartum period was induced by the acceleration of the autoimmune reaction by the same mechanism of postpartum autoimmune thyroiditis.

Adult↗
PubMed1988

[Serum thyrotropin concentrations in patients with Sheehan's syndrome: reevaluation of the usefulness in the diagnosis].

Serum concentrations of thyroid-stimulating hormone (TSH) were determined, using a highly sensitive immunoradiometric assay (IRMA), in 10 patients with Sheehan's syndrome. Serum TSH levels in these patients were from 2.3 to 9.0 microU/ml, with the mean of 6.4 +/- 2.3(SD) microU/ml, and the data were similar to those measured by a conventional RIA method. The levels of serum TSH in these patients were normal or even higher than those of healthy women (1.8 +/- 1.3 microU/ml). After supplement therapy by cortisol, serum TSH levels decreased, but remained within the detectable range that was greater than 0.15 microU/ml. After supplement therapy by 1-thyroxine (T4), serum TSH levels moderately decreased in all patients, and excessive 1-T4 administration resulted in a fall of serum TSH levels to lower than the detectable limit. Thyroidal 123 I uptake was low in 3 out of 6 patients examined, which supports a recent hypothesis of reduced biological activity of the patient's TSH. While, the remaining 3 patients had normal thyroidal 123 I uptake, and administration of perchlorate had no effects on thyroidal radioactivity. Thus, it may be possible that in the former group of patients the TSH has a reduced biological activity, and in the latter group of patients, iodine trapping is intact but further synthesis and secretion of thyroid hormone from the gland are impaired. When the response of serum TSH to TRH was examined, the peak serum TSH levels increased in all patients. The peak TSH levels were 4.8 to 10.2 microU/ml with the mean of 7.5 microU/ml, but the data overlapped with those of normal subjects (peak level ranging 5.9 approximately 27.7 microU/ml).(ABSTRACT TRUNCATED AT 250 WORDS)

Adult↗
PubMed1988

Development of hyperthyroidism following primary hypothyroidism: a case report with changes in thyroid-related antibodies.

We report a 48-year-old woman who developed hyperthyroidism following primary hypothyroidism. The serum T4 level was initially low and serum TSH level was high with clinical signs of hypothyroidism. The thyroid gland was not enlarged. Therapy with L-T4 was started. Three years later she developed hyperthyroidism; serum free T4 increased to 29.1 pmol/l after cessation of L-T4 therapy. The 123I thyroid uptake was increased with no suppression by exogenous T3. When she was hypothyroid, the activity of thyroid stimulating antibodies (TSAb) in serum measured by cyclic AMP production in cultured porcine thyroid cells were negative at 93.4% (normal less than 140%), while thyroid stimulation-blocking antibodies (TSBAb) determined by inhibition of TSH-induced cyclic AMP increase were positive at 96.1% (normal less than 40%). When hyperthyroidism subsequently occurred, TSBAb became negative (30.9%), while TSBAb became positive (163.3%). The findings indicate that hypothyroidism due to the potent TSBAb activity is not always persistent, but can be changed when various types of thyroid-relating antibodies change in the course of the disease.

Antibodies↗
PubMedDate not supplied

Treatment with rhG-CSF for osteomyelitis in a patient with p47-phox-deficient chronic granulomatous disease.

A 24-year-old woman with osteomyelitis was diagnosed as having p47-phox-deficient chronic granulomatous disease (CGD). The patient showed a marked deficiency of p47-phox, which is very rare in Japan. As the clinical response to various antibiotics including sulfamethoxazole-trimethoprim was not satisfactory, we added recombinant human granulocyte colony-stimulating factor (rhG-CSF) to the treatment protocol. We report the beneficial clinical course of the patient, together with the effect of rhG-CSF on the granulocyte function, and the present report indicates that rhG-CSF is useful for the treatment of antibiotic-resistant infection in the variant type of p47-phox-defective CGD.

Adult↗