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Volume 73 (1997)
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Volume 73, Issue 6
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[in Japanese]1997 Volume 73 Issue 6 Pages 591-596
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSDownload PDF (652K) -
Kayo YAMANAKA1997 Volume 73 Issue 6 Pages 597-609
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSGrowth hormone (GH) and IGF-I are considered to be essential to adipocyte differentiation and may play a key role in fat mass accumulation in vivo. The aim of this study is to determine the effect of high lipid content diet (HL) on serum GH and tissue IGF-I mRNA levels in rats. Male Wistar rats were administered a diet containing 29% energy as lipid for 18 weeks. Significant body weight increase was observed in HL-treated rats at 28 weeks of age, comparing with normal control (CO) rats. Significantly increased fat accumlation was also shown in epididymal and subcutaneous adipose tissues in HL rats. Serum GH levels in HL rats were significantly lower than those in CO rats (HL=16.5±2.9 (SEM)ng/ml vs CO=31.7±6.0, p<0.01). Total RNA was extracted and applied to northern blot to hybridize with a rat IGF-I cDNA probe. IGF-I mRNAs were expressed at 7.5 kb with several minor bands in liver and white adipose tissue. Hepatic IGF-I mRNA/β-actin mRNA ratio in HL rats was significantly decreased (HL=0.74±0.07 vs CO=1.66±0.59, p<0.01) in accordance with decreased serum GH levels. The epididymal fat IGF-I mRNA/β-actin mRNA ratio in HL was also significantly lower than those in CO (HL=0.56±0.13 vs CO=0.81±0.14, p<0.02), whereas there was no significant difference in subcutaneous fat IGF-I mRNA/β-actin mRNA ratio between HL and CO (HL=1.61±0.31 vs CO=1.68±0.42, NS). IGF-I mRNA levels in both liver and epididymal fat in HL rats decreased proportionately to serum GH levels, suggesting that hepatic and epididymal fat IGF-I mRNA levels are mainly regulated by GH. In an obese state, increased visceral fat accumulation may be promoted by decreased serum GH levels or vice versa.View full abstractDownload PDF (1479K) -
Jinchi KIM, Zen-O YAMADA, Soitsu FUKUCHI, Yasuo FUKAYA, Hironobu SASAN ...1997 Volume 73 Issue 6 Pages 611-615
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSA rare case of primary aldosteronism with two clear cell adnomas and one black adenoma in the left adrenal gland is reported. All of these adenomas were almost the same size about 10 mm in diameter.
A 50-year-old woman was admitted to our hospital complaining of hypertension, hypokalemia (3.2 mEq/l) and hyperglycemia. Plasma aldosterone level was high (280 pg/ml), while renin activity was suppressed (0.1 ng/ml/h). Plasma cortisol level was normal high (18.8μg/dl) and urinary 17-OHCS was within normal range. Abdominal CT revealed a left adrenal tumor.
The left adrenal gland was completely removed. There were two clear cell adenomas and one black adenoma. There were 20 percent compact cells in one of the clear cell adenomas, while the other consisted with only clear cells. Black adenoma consisted of eosinophilic cells with infltration of lipofustin granules.
Immunohistochemical analysis was done on each adenoma and the adjacent adrenal tissue. P450-SCC, 3βHSD, P450-C21, C11, C17 and DHEA-ST were all detected in the black adenoma, while P450-C17 and DHEA-ST were not present in the clear cell adenomas. DHEA-ST was suppressed in the adjacent adrenal tissue.
It is suggested that excessive aldosterone was secreted autonomously in only the the clear cell adenoma accompanying compact cells, and that the other adenomas were non-functioning.View full abstractDownload PDF (1079K) -
Megumi MATSUNO, Motoi SOHMIYA, Masateru NISHIKI, Satoshi OHGUNI, Yuzur ...1997 Volume 73 Issue 6 Pages 617-622
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSLevothyroxine sodium (Thyradin S®) (L-T4) is one of the most useful drugs for replacement therapy in primary hypothyroidism. There have been few side effects reported. We report a rare case with chronic thyroiditis accompanied by drug eruption induced by L-T4.
A 68-year-old man consulted our hospital for nasal bleeding and anemia. He was diagnosed as allergic rhinitis and hypothyroidism due to chronic thyroiditis, and the replacement with L-T4 was started. On the eleven months after the start of L-T4 treatment, erythematous eruption with severe itching appeared on his chest, back, upper and lower extremities. In spite of stopping all other drugs except L-T4, the eruption did not disappear. When the L-T4 was discontinued, the eruption disappeared within 3 weeks. The scratch patch test revealed the hypersensitivity for L-T4 in this patient. The challenge test with L-T4 resulted in itching but not eruption after 72 hours. The skin biopsy revealed the superficial perivasculitis accompanied by infiltration of eosinophils and lymphocytes. These findings suggest that the drug eruption was induced not only by L-T4 accumulation, but partly attributed to allergic reaction for L-T4.View full abstractDownload PDF (1330K) -
Kuniki EGUCHI, Kazunori ARITA, Kaoru KURISU, Tohru UOZUMI, Atsushi TOM ...1997 Volume 73 Issue 6 Pages 623-627
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSWe describe a patient who developed panhypopituitarism due to a Rathke cleft cyst. The patient, a 63-year-old man with a 25-year history of diabetes insipidus was presented with obtunded consciousness. On admission, his serum Na level was 119 mEq/l. MRI revealed a intra- and supra-sellar hyperintense cyst on T1 and T2 weighted images. The cyst wall was not enhanced by Gd-DTPA. Endocrine studies revealed the existence of panhypopituitarism. The cyst contents and part of the wall were removed using the transphenoidal approach. Cyst wall was found to consist of a single layer of ciliated columnar cells on microscopic examination.
Hypopituitarism, visual disturbance and headache are common symptoms in patients with the symptomatic Rathke cleft cyst. The incidence of hypopituitarism reached 30-39% of the patients with symptomatic Rathke cleft cyst. Few reports on panhypopituitarism accompanied with Rathke cleft cyst have been published. In our case, the long period between the onset of diabetes insipidus as an initial symptom and surgery is thought to be the major cause of panhypopituitarism because the pituitary dysfunction has arisen cumulatively during 25-year period.
In conclusion, a patient with intra- and/or supra-sellar cystic lesion should be investigated with neurological and endocrinological examinations including hypothalamo-pituitary function tests, and managed appropriately. In patients without any symptoms and signs, regular follow-up is needed with attention to the new endocrinological and neurological deficit besides abscess formation in the cyst and hemorrhage into the cyst.View full abstractDownload PDF (1021K) -
Shin-ichi UMEZAWA, Akira KANAMORI, Shin-ichi NAKAJIMA, Yasuyuki JIN, K ...1997 Volume 73 Issue 6 Pages 629-636
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSFamilial isolated hyperparathyroidism (FIHP), a specific type of primary hyperparathyroidism, is known to be asymptomatic despite the accompanying dangerously high levels of calcium and advanced bone or renal disease. In this report, we describe the case of a 47 year-old female with familial hyperparathyroidism, being presented in hypercalcemic crisis with severe bone lesions. She complained of the classic symptoms of hyper-calcemia, such as anorexia, thirst, palpitation and muscle weakness. A physical examination revealed a hump-back deformity associated with a severe thoracic scoliosis and a large neck tumor at the upper pole of the left thyroid lobe. Her serum calcium concentration was 19 mg/dl, her alkaline phosphatase concentration was 2,691 IU/L, and her intact PTH was 3,830 pg/ml. A skeletal survery of the whole body showed the severe osteolytic changes of hyperparathyroidism. Cervical exploration was undertaken, and a tumor (6cm in diameter) was removed. Histological examination revealed a parathyroid chief cell adenoma. In review of this case along with previously reported cases of FIHP in Japan, all cases of hypercalcemia with a calcium concentration greater than 14 mg/dl had exhibited typical hypercalcemic symptoms. Also, many of the urolithiasis-negative cases had significant bone symptoms and signs associated with hyperparathyroidism. Age at diagnosis as hyperparathyroidism is higher in bone-type cases than in urolithiasis-positive cases. The reason for these results is probably due to a lack of urolithiasis that may show early signs of hypercalcemic disorder. In conclusion, it is suggested that hypercalcemic and bone symptoms associated with hyperparathyroidism are not rare in patients with FIHP in Japan, and early recognition of hyperparathyroidism in the family member through long-term follow-up is necessary to avoid a life threatening hypercalcemic crisis as in the present case.View full abstractDownload PDF (1766K) -
Shigeki KOIZUMI, Mutsuo BENIKO, Akemi IKOTA, Hiromitu YAMAMOTO, Akira ...1997 Volume 73 Issue 6 Pages 637-642
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSA 50-year-old man attended our hospital with the chief complaint of osteoporosis. He was diagnosed with diabetes mellitus at 32 years of age and was treated with 3.75 mg of glibenclamide. Physical examination showed short statue with BMI of 25.3 kg/m2, sparse body hair, gynecomastia, and bilateral small testes. Endocrinological studies showed that basal levels of LH and FSH were elevated and the serum testosterone level was low. An impaired response of testosterone to HCG was observed. These results indicate hyper-gonadotropic hypogonadism. Cytogenetic studies showed 46,XX karyotype with unidentified region on p22 of an X chromosome. Sex determining region of the Y chromosome (SRY) was detected by PCR technique. The diagnosis of XX-male was made from the above findings. He had a fracture in the 3rd lumbar vertebrae and severe osteopenia (bone mineral density of 0.72g/cm2) by dual energy X-ray absorptiometry. Hypogonadism is a well recognized cause of osteoporosis in male, in which testosterone deficiency may cause a bone deficit in those patients. XX-male may be another hypogonadism that leads to osteoporosis in male. Forty-eight cases of XX-male were reported in Japanese literatures. The present case is the first case of XX-male with osteoporosis and the second case with diabetes mellitus.View full abstractDownload PDF (1183K) -
Noritaka IWATANI, Takako OTSUKA, Hiroyo MABE, Mihoko KODAMA, Teruhisa ...1997 Volume 73 Issue 6 Pages 643-649
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSWe describe the patient of a 23-year-old female with Laron syndrome who was the first case of GH-receptor gene mutation detected in Japan. Her height and weight were 125.0 cm (-6.6 SD) and 56.0kg. Her father and one of her three brothers were 158 cm (-2.2 SD) and 160 cm (-1.9 SD) in height, respectively, but her mother and remaining two brothers were of normal height. Serum growth hormone (GH) concentration was high in basal levels (9.6-23.6ng/ml) and reached extremely high levels responding to the provocative tests of insulin hypoglycemia and arginine infusion (60.8 and 104.8ng/ml, respectively). Serum IGF-I concentration was low (42ng/ml), serum GH-binding protein level was undetectable at less than 1.16ng/ml, and serum IGF-binding protein-3 level was 0.51μg/ml (normal: 2.59-4.26).
Each exon and flanking part of intron of the GH-receptor gene from the genomic DNA by PCR was sequenced. A G to A mutation was identified at the first position of intron 4. This mutation was homozygous and could possibly cause an abnormal splice site resulting in a production of abnormal GH-receptor protein.View full abstractDownload PDF (694K) -
Study by Dual Energy X-ray Absorptiometry (DEXA)Masanori KANATANI, Toshitsugu SUGIMOTO, Yasuhide TOKKODA, Kazuo CHIHAR ...1997 Volume 73 Issue 6 Pages 651-658
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSThe effects of long-term treatment with 1α3-hydroxyvitamin D3[1α(OH)D3] on bone mineral density, fracture incidence and chemical parameters in postmenopausal osteoporosis were examined.
Thirty two postmenopausal women were divided into 2 groups: Group 1 (n=10) as the control group and Group 2 (n=22) which was given 1μg of 1α(OH)D3 per day without supplemental calcium for two to three years.
Lumbar (L2-L4) bone mineral density (BMD) determined by dual energy X-ray absorptiometry was maintained for the first year and increased two and three years after the treatment with 1α(OH)D3, while BMD of controol group decreased yearly for three years. Percent change of corrected L2-L4 BMD increased by 2.55% with 1α(OH)D3 treatment and decreased by 1.32% with the control group after two years. This difference was significant (P=0.014). In all younger postmenopausal patients (55-65 years old) except for two patients, 1α(OH)D3 treatment increased the percent change of L2-L4 BMD.
The vertebral fracture rate in the treated group was 39/1000 patient years and that in the control group was 111/1000 patient years although there was no significant difference in fracture rate between them. There were no patients who showed hypercalcemia nor an abnormally high serum creatinine level in both groups during the study period. These results indicate that 1α(OH)D3 treatment is effective for increasing lumber BMD without any serious adverse effects in postmenopausal osteoporosis.View full abstractDownload PDF (788K) -
Ken KATOH, Sanae MIDORIKAWA, Koichi ASAHI, Daishiro YAMADA, Minoru KUR ...1997 Volume 73 Issue 6 Pages 659-666
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSTo investigate the acute effects of insulin on the fibrinolytic system in essential hypertension (EH), plasma plasminogen activator inhibitor 1 (PAI-1), which is a major anti-fibrinolytic substance, tissue plasminogen activator (tPA), thrombomodulin (TM), triglycerides (TG), and free fatty acids (FFA) were measured in 14 middle-aged, non-obese patients with EH and 6 age-matched control subjects at a hyperglycemic, euglycemic glucose clamp before and after a 2 hour-insulin infusion.
Serum insulin level during the clamp was 78-186μU/ml and blood glucose was maintained at 80-105mg/dl. The glucose infusion rate was significantly less in the patients (5.3±1.7 mg/kg/min) than in the controls (9.1±1.3mg/kg/min). During the clamp, plasma PAI-1 concentration did not significantly change in the patients whereas it was significantly lowered in the controls (by 42%): abnormality of regulation of PAI-1 is related to the degree of insulin resistance as indexed by lower glucose infusion rate in some of them. There was no suppression of tPA and TM both in the patients and controls. In contrast, TG and FFA levels were significantly suppressed by the insulin infusion in both groups.
We conclude that insulin acutely suppresses serum TG and FFA, but not plasma PAI-1 in patients with EH, implying that the acute effects of insulin on the lipids and PAI-1 are independent of each other. The findings suggest that the abnormal regulation of PAI-1, but not lipid, exists in patients with EH. This abnormality may well be related to the initiation and/or evolution of vascular complications in EH.View full abstractDownload PDF (670K) -
Hiroyuki GOTO, Yasuhiro SUMIDA, Kazuya MURATA, Atsushi EZAKI, Hitoshi ...1997 Volume 73 Issue 6 Pages 667-673
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSThiamazole is well known for treatment of Graves' disease, and thiamazole-associated liver injury occurs very rarely. We report a case of hyperthyroidism complicated with drug-induced cholestatic liver injury during treatment with thiamazole.
A 26 year-old male visited our hospital on October 3, 1991 because of mild fever, loss of weight and general fatigue. Because of diffuse goiter and laboratory data indicating hyperthyroidism, a diagnosis of Graves' disease was made, and administration of thiamazole was started on January 9, 1992. From January 20, fever, white watery stool and jaundice developed in a few days, and he was admitted to our hospital on February 3.
Physical examination showed jaundice and goiter, and the blood chemistry test indicated liver injury (GOT 111 IU/1, GPT 196 IU/1, T. Bil 24.3 mg/dl). Viral markers of hepatitis A, B or C virus were negative. An echo examination of the abdomen showed no dilatation of bile ducts. Liver biopsy specimen indicated cholestasis without infiltration of inflammatory cells.
Drug-induced cholestatic liver injury was suspected, and administration of thiamazole was stopped. Afterward, the serum transaminase levels decreased, but jaundice remained. Administration of prednisolone resolved jaundice.View full abstractDownload PDF (1250K) -
Keiichi KAMEOKA, Haruhiko ISOTANI, Keizo FURUKAWA1997 Volume 73 Issue 6 Pages 675-681
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSSince acute-onset insulin-dependent diabetes mellitus (IDDM) usually demonstrates a greater number of HLA antigens related to IDDM than slowly progressive IDDM (SPIDDM), the type of diabetes may be affected by a dosage effect of HLA antigens. The purpose of this study is to elucidate the dosage effect of HLA antigens in modifying the type of diabetes. We assayed antibodies to glutamic acid decarboxylase (GADab) in 253 non-insulin-dependent diabetes mellits (NIDDM) patients and 17 IDDM patients. We assayed HLA antigens (class I A, B, C antigens, class II DR, DQ antigens) in 7 of 8 GADab-positive NIDDM patients and 3 GADab-positive acute-onset IDDM patients, whose disease etiologies may be related to the background of autoimmunity. We tried to score the association of HLA antigens with IDDM as positive or negative in each patient. If the patients had one HLA antigen related to IDDM, they were given one point. If they had one HLA antigen showing a negative association with IDDM, one point was subtracted from the score. HLA scores of GADab-positive patients with NIDDM tended to have a lower score, but exceptionally there were a few higher HLA scores among NIDDM patients. Both GADab-positive and negative IDDM patients tended to have higher HLA scores.
Our results suggest that measurement of HLA antigens as well as GADab can be of clinical value in predicting the type of diabetes. Moreover, it may be possible to detect patients who should receive insulin during the early stage of GADab-positive NIDDM.View full abstractDownload PDF (657K) -
Hiroyuki HOSOJIMA, Kenzo UCHIDA, Kazuko KITAGAWA1997 Volume 73 Issue 6 Pages 683-691
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSHereditary crystalline corneal dystrophy of Schnyder, in which cholesterol or cholesterol ester deposit in the centers of the bilateral corneas, is rare, and co-existed frequently with familial hyperlipidemia and/or hyperlipoproteinemia, Xanthelasma or active genu valgum. We report two cases of Graves' disease and another three subjects in a family who have abnormal thyroid hormone concentrations accompanied with Schnyder's corneal dystrophy.
Case 1: A 48-year-old man was referred for studying the relation between the corneal turbidity and the hormonal dysfunction. He had noticed a decreased bilateral visual acuity several years ago. He was diagnosed with Schnyder's corneal dystrophy because of deposits of white crystalline-like substances in his corneas. Both a son and two daughters had similar opaque corneas with in a few years. From last year, he had a fine hand tremor, but no difficulties in his working. His serum levels of T3, free T4 (FT4) and TSH were 3.5 pmol/L, 39.7 pmol/L and less than 0.4 mU/L, respectively, and TRAb was positive. He was diagnosed with Graves' disease, and then treated with an oral anti-thyroid drug (300 mg/d of Methimazole).
Case 2: A 14-year-old girl (the eldest daughter of the subject in Case 1) developed the bilateral Schnyder's corneal dystrophy within a few years. When her father began treatment for Graves' disease, her thyroid hormone concentrations were almost within normal ranges except for less than 0.4 mU/L of TSH. One year later, she suddenly had a palpitation, nausea and a swelling of the anterior neck, and was referred to a pediatric hospital. Her serum levels of T3 and FT were 3.3 pmol/L and 75 pmol/L, and TRAb and TSAb were positive. She was also diagnosed with Graves' disease and then treated with an anti-thyroid drug (150 mg/d of PTU). In his family, the father, a son and two daughters had Schnyder's corneal dystrophy, and co-existing genu valgum, congenital coxal luxation, or hyperlipoproteinemia. The Graves' disease was occurred in Case 1 and 2, and the other cases had partly elevated concentrations of thyroid hormones and TSH level. The cases of Graves' disease or other thyroid disease accompanying with hereditary Schnyder's corneal dystrophy, however, have not been reported. Although they had similar HLA typings, A24 (9) in locus A, BW52 (5) or BW61 (40) in locus B, CW3 in locus C, and DR9 in locus DR, no specific TSH receptor gene was found in all subjects.
This is a first report of the occurrence of Graves' disease with the hereditary Schnyder's corneal dystrophy.View full abstractDownload PDF (1050K) -
Report of 14 casesMasako KANBE, Takao OBARA, Kiyomi YAMAZAKI, Kenzo HIROSE, Takahiro OKA ...1997 Volume 73 Issue 6 Pages 693-700
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSA retrospective review of 396 patients with primary hyperparathyroidism (pHPT) treated surgically revealed a past history of neck external irradiation in 14 patients (3.5%). The mean interval between radiation exposure and the diagnosis of pHPT was 41 years (range, 31-49 years). All of the patients were women who had been exposed to radiation more than 31 years before. There was a significant difference in the male: female ratio of affected patients (P<0.001). The parathyroid histology was adenoma in 13 patients and carcinoma in one. In ten patients, normal parathyroid gland biopsies were undertaken. Among normal parathyroid glands from two patients, there were nodular lesions. Thirteen (93%) of the 14 patients who had undergone neck irradiation had thyroid nodular disease. In contrast, only 98 (26%) of the 382 patients who had not undergone neck external irradiation had thyroid nodular disease, and the difference between the two groups was significant (P<0.001).
Neck irradiation has been shown to increase the risk of parathyroid and thyroid nodular diseases. If neck exploration is necessary in a patient who has received neck irradiation, both the thyroid and parathyroid glands should be carefully evaluated before and during surgery.View full abstractDownload PDF (1068K) -
Takashi KOMAKI, Kouji YOSHINO, Noriyoshi YAMAKITA, Kiyoshi MIURA, Shin ...1997 Volume 73 Issue 6 Pages 701-708
Published: November 20, 1997
Released on J-STAGE: September 24, 2012
JOURNAL FREE ACCESSA case of adult-onset hypophosphatemic vitamin D resistant osteomalacia is reported. A 45-year-old female who complained of lumbar pain was admitted to our hospital in December 1994. She was normal stature and had no stigmata of rickets. There was no family history of osteomalacia or rickets. She had severe hypophosphatemia (1.1 mg/di), high concentration of serum alkaline phosphatase (486 IU/l), decreased plasma 1,25-(OH)2 vitamin D concentration (lower than 5.0 pg/ml), high-normal parathyroid hormone concentration (560 pg/ml by double antibody RIA, and 48 pg/ml by IRMA), decreased tubular reabsorption of phosphate (% TRP: 75.2%), and decreased tubular maximum transport of phosphate per glomerular filtration rate (TmP/GFR: 0.83 mg/dl). Looser's zone was observed in pubis and ischium on radiograph. The 99mTc-MDP bone scintigram showed an evidence of increased bone turnover. Clinically, there was no proximal muscle weakness. She was treated with oral administration of 1,25-(OH)2 vitamin D3 (1-6μg, daily) and phosphate supplement (1.0-0.3g, daily) resulting in slow clinical, and biochemical improvement.View full abstractDownload PDF (1235K)
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