Saccharaed ferric oxide (SFO)-induced osteomalacia develops when excessive SFO infusions are administrated to patients with anemia for prolonged periods for a few years. The small particles and almost neutral saccharide of SFO filter through the glomerular tufts into the renal tubules, resulting in impairment of proximal renal tubular function, particularly renal reabsorption of phosphate and 1α-hydroxylase activity, resulting in decreased serum levels of phosphorus and active vitamin D, both of which lead to development of hypophosphatemic osteomalacia. Furthermore, SFO, at concentrations ttainable in serum, exacerbates the osteomalacia by inhibiting bone formation directly. In contrast to itai-itai disease, another iatrogenic osteomalacia due to cadmium nephropathy [44], the proximal renal tubular function impairment induced by SFO is reversible simply by discontinuing the nephrotoxin, which is followed by improvement of all the clinical manifestations, except bone deformities.
So far, SFO-induced osteomalacia, that is, SFO-induced osteopathy due to nephropathy, has been reported only in Japan, probably due to the lax surveillance system of the health insurance scheme. All physicians who prescribe SFO should be aware of its severe adverse effects. We hope that such iatrogenic osteomalacia caused by abusive infusion of SFO will never again be reported in our country.

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The endocrine function and pituitary imaging in Sheehan's syndrome more than 30 years after causative events were evaluated. Magnetic resonance imaging (MRI), a combined anterior pituitary test, plasma vasopressin-to-osmolality adaptation study, and antithyroid and antipituitary cell antibody measurement were performed in 6 women with Sheehan's syndrome. The interval from delivery to the onset of symptoms of hormonal deficiency ranged from 3 to 32 years. Since clinical onset, all had received glucocorticoid and thyroid replacement therapy. Cranial MRI examination showed an “empty sella” in 5 cases. Among these, 2 of 5 (40%) demonstrated panhypopituitarism and the other 3 (60%) maintained gonadotropin response. The pituitary gland was normally discernible but with a low- intensity lesion on T1-weighted images in a patient who maintained PRL and gonadotropin responses. Posterior pituitary function was abnormal in 3 of 6 (50%) despite the absence of polyuria. No antipituitary antibodies were detected in any of the cases. Thyroid peroxidase antibody was negative in all cases, but antithyroglobulin antibody was detected in 2 of 6 (33%). Thyroid-stimulating antibody was not detected, but one case had an anti-TSH antibody. Thirty years after the initial events, most patients with Sheehan's syndrome showed signs of an empty sella on MRI, all having noticeable suppression of anterior and/or posterior pituitary hormones with no related autoimmunity.

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The Foundation for Growth Science has been controlling the use of GH by its registration system, which includes a scoring system for the eligibility for GH treatment according to the diagnostic criteria for GH deficiency (GHD) established by the Study Group for Hypothalamo-pituitary Disorder of the Ministry of Health and Welfare. Until 1995, 28, 876 patients with GHD (19, 432 boys and 9, 444 girls) had been registered as eligible for GH treatment. The number of patients registered in a year increased gradually till 1990 due to the unlimited hGH supply by recombinant techniques and the change in the criteria for GH treatment and the number registered became stable after 1990. The frequency of GH- treated patients is calculated to be 55.2/100, 000 persons (72.2/100, 000 in boys and 37.1/100, 000 in girls) in patients born between 1960 and 1990. The highest frequency was 148.4/100, 000 persons (191.7/ 100, 000 boys and 103.7/100, 000 girls) in 1981, when 2, 278 patients (1, 508 boys and 770 girls) were born. Eligibility for GH treatment is assessed according to the scoring system which is basically dependent on peak GH values in provocation tests so that standardization of GH values measured with the various commercial GH kits is required to avoid inequality of patients' access to the treatment. In samples obtained by GRF test in 10 normal volunteers, hGH was measured with seven human GH (hGH) kits at a laboratory center. Since the RIA value has been used historically for the diagnosis of GHD, the mean of two RIA measurements was selected as the basis for the standardization procedure and the linear regression formula was used for each hGH kit. After the freely available supply of hGH obtained by recombinant DNA techniques, the role of the Foundation for Growth Science has changed to avoid hGH abuse. Even with this regulation, the frequency of registered patients may indicate a tendency to GH overuse.

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The cDNA of the common α-subunit of human glycoprotein hormone was mutated by site directed mutagenesis in the CMGCC region composed of cysteine-methionine-glycine-cysteine-cysteine (position 28-32). The cDNA of wild-type human thyrotropin (hTSH) β-subunit and that of wild-type or mutant common α-subunits were co-transfected into COS-I cells. The concentration of hTSH determined by two immunoradiometric assay systems was detectable in culture media of COS-I cells transfected with wild-type (CMGCC) and a mutant (CMGCC) α-subunits but not four other mutants (YMGCC) (CMRCC) (CMACC) (CMGCC). The present data with the other studies on wild-type or mutant glycoprotein hormones support our hypothesis that an amino acid motif of “C-X-G-X-C” in the common α-(CMGCC in human) and β-(CAGYC in human) subunits play an important role in biosynthesis of glycoprotein hormones in all species.

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We performed a long-term (35 to 45 year) follow-up study on patients who underwent surgery for intrathyroidal papillary thyroid carcinoma in order to reveal the natural history of the disease. Forty-nine patients underwent primary surgery for intrathyroidal papillary carcinoma during an 11-year period, 1950-1960. Various primary surgeries were performed, including neck exploration alone, tumor enucleation, hemi-, subtotal- and total-thyroidectomy in 2, 7, 21, 5 and 14 instances, respectively. Postoperative external irradiation was performed for most patients during the latter half of the period, and TSH suppression was carried out from 1956 through 1970. Follow-up studies were done in 1958, '62, '66, '69, '76 and currently 1997. Two patients, who had had only neck exploration and external irradiation, subsequently spent nearly normal lives for 10 and 32 years. Of the 28 patients who received hemithyroidectomy or lesser surgery, cancer recurrence in the remnant thyroid occurred in nine, of whom five received reoperation. At present, of all 49 patients, 22 are alive and well, and three are alive with asymptomatic recurrence. Only one male patient who had noted the initial lymph node metastases at age 15 died of bone metastasis 22 years after neck surgery. No other patients died definitely of thyroid cancer, although the causes of three deaths were unknown and one patient was lost after incomplete resection. The results of this study strongly support the idea that the majority of intrathyroidal papillary carcinomas remain non life-threatening for over 40 years and that they can be successfully treated by complete removal of macroscopic tumors by conservative surgery, hemi- or subtotal thyroidectomy, without associated adjuvant therapies.

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A 74-year-old man developed Cushing's syndrome and hypokalemia due to ACTH-independent bilateral macronodular adrenocortical hyperplasia (AIMAH) with excessive secretion of mineralocorticoid hormones. Plasma concentrations of weak mineralocorticoids were high. The increase in plasma cortisol did not have a diurnal rhythm, and was not suppressed by a high dose of dexamethasone. Plasma ACTH was undetectable, but plasma cortisol was increased by ACTH administration. The concentrations of mineralocorticoids, especially deoxycorticosterone and corticosterone were increased, and augmented the response to ACTH administration. Plasma renin activity and aldosterone concentrations were rather suppressed. Both adrenal glands, with a total weight of 110g, were enlarged and contained several macronodules. These nodules were composed of hyperplasia of small cortical cells and usual clear cells. This is a rare case of ACTH-independent bilateral macronodular adrenocortical hyperplasia because there were excessive secretion of mineralocorticoid and hypokalemia.

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To determine whether nonclassical steroid 21-hydroxylase deficiency in Japan has the same molecular basis as in western countries, we have characterized the mutations of the CYP21 gene in 7 Japanese patients with nonclassical (NC) steroid 21-hydroxylase deficiency. In the Japanese NC cases the P30L was present in one allele in 5 of the 7 patients and on both alleles in one patient. By contrast, the V281L mutation, which was present in about 60% of NC cases in western countries, was not identified in any patient. Among our 7 cases, 4 were detected through neonatal mass screening by a mild increase in serum 17-hydroxyprogesterone (without any symptoms of CAH) at birth, but the 2 cases who were diagnosed as adults were born before nationwide neonatal screening was instituted, so that the Japanese neonatal screening program does detect some cases of NC steroid 21-hydroxylase deficiency. We suggest that P30L mutation is more frequent in Japanese NC CAH than V281L and that the frequency of the mutations causing NC steroid 21-hydroxylase deficiency in Japan might be different from that in western countries.

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We encountered a case of a pregnant woman with adrenal causes of Cushing's syndrome who exhibited congestive heart failure as an initial symptom. Since the patient was also a diabetic, we treated her with high levels of diuretics and insulin. Echocardiography revealed a remarkable thickening of the left ventricle without asymmetric hypertrophy. The diagnosis of Cushing's syndrome caused by adrenal adenoma was confirmed by the endocrinological data and magnetic resonance imaging. The right adrenal adenoma was removed in the 28th week of pregnancy. After the operation, her congestive heart failure and hyperglycemia dramatically improved. Five weeks after the operation, she delivered a normal infant by caesarean section without complications. Only 4 months after delivery, the thickening of her left ventricle was normalized. We consider that the progression of her left ventricular hypertrophy induced by the changes in hemodynamic load during pregnancy may have been augmented by the excess of plasma cortisol. Operative therapy may be recommended for pregnant Cushing's syndrome patients with severe hypercortisolism complicating congestive heart failure.

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In order to compare the bone aging process in Werner's syndrome, a disease characterized by premature aging, and that in natural aging, we have assessed the bone status in a total of 19 cases (11 males and eight postmenopausal females) with Werner's syndrome. The spinal deformity index was determined for a total of 87 vertebral bodies from nine male patients and for a total of 61 vertebral bodies from six female patients. In the male patients, 15 vertebral deformities among 87 vertabrae in seven patients were observed, and the incidences of patients bearing deformity and deformed vertebrae were 77.8 (7/9) and 17.2% (15/87), respectively. In the female patients, the corresponding incidences were 50.0% (3/6) and 4.9% (3/61), respectively. The χ²-values for the incidences of patients bearing deformity and deformed vertebrae in the male and the female patients were 1.25 (P=0.26) and 5.24 (P<0.03), respectively. The metacarpal cortical thickness (MCI) was also measured on hand X-ray films, and the Z scores for MCI in the male (n=9) and female (n=3) patients were -0.8±0.3 and -1.6±0.3, respectively. This gender-based difference in the MCI was not statistically significant but, the Z scores for MCI in both the males and the females were significantly smaller than those of corresponding age-matched controls (P<0.05 and 0.01, respectively). The serum levels of calcium, intact PTH, 25-OH vitamin D and 1, 25(OH)₂ vitamin D in three male and three female Werner's syndrome patients were within the reference ranges with one exception. The serum intact osteocalcin levels in the male (2.2±0.6 ng/ml) and female (2.6±0.8ng/ml) patients were lower than the age-matched control values (P<0.05 for males) and the postmenopausal control values (P<0.05 for females). In conclusion, the incidence of spinal bone deformity was higher in males with Werner's syndrome than that in female patients. The cortical bone thickness decreased in both the male and the female patients. A low serum intact osteocalcin level was a characteristic finding in the male and in post-menopausal Werner's syndrome, so that the bone changes such as vertebral fracture incidence and the serum intact osteocalcin level in Werner's syndrome may differ from those in normal aging in the general population.

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In order to determine whether thyrocyte responsiveness to TSH is correlated with post-operative thyroid status, thyrocytes obtained from 51 patients with Graves' disease were cultured for 3 days. This was followed by culture with 10mU/mL of TSH (TSH group) or without TSH (control group) for 3 additional days. On the 8th culture day, the amount of thyroglobulin (Tg) secreted into the culture medium was assayed and the Tg ratio (amount of Tg from the stimulated group/amount of TG from the control group) was calculated. Log₁₀ (the Tg ratio) was used for the statistic study. The post-operative status of the patient was determined based mainly on the serum concentration values of free T3 and T4 obtained between 5 and 16 months after surgery. Multivariate analysis revealed that among age, gender, TSH-binding inhibitory immunoglobulin (TBII) obtained before the surgery, estimated weight of the thyroid gland remnant, and Log₁₀ (Tg ratio), only Log₁₀ (Tg ratio) was statistically significant (P=0.008). When the Log₁₀ (Tg ratio) value was small, most patients showed signs of as post-operative hypothyroidism, and as the value became larger as the number of euthyroid and/or hyperthyroid patients increased.

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The temporal changes in excitability in ventromedial hypothalamic neurons which project to the midbrain central gray caused by chronic estrogen exposure were measured in ovariectomized rats. In 66 urethane-anesthetized animals which had either no capsule, or had a capsule containing estrogen implanted for 24h, 48h, 72h or 96h, 189 antidromic action potentials were recorded in the ventromedial hypothalamus (VMN) in response to stimulation of the midbrain central gray (CG). The antidromic activation threshold was lower in 40 recordings made from the VMN in the 72-h or 96-h group than in 67 from the no-treatment, 24-h or 48-h group. The decrease in the threshold coincided with the emergence of the lordosis reflex. In a separate cohort of ovariectomized rats, any absence of estrogen for 2-48h before and during the measuring abolished the neuronal as well as the behavioral effects at 72h. The results suggest that the lordosis reflex induced by estrogen alone depends on the excitation of VMN efferents to the CG. In addition, the indispensability of estrogen at the time of electrophysiological and behavioral evaluation indicates that a certain direct neural action of estrogen is involved in the behavioral activation.

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Bromocriptine (BC) is now an accepted primary therapeutic agent for patients with microadenoma. But, for patients with large or giant prolactinomas, the treatment choice is controversial. This report focuses on long-term results of the BC effect on 10 giant prolactinomas (maximum diameter more than 40mm and the serum PRL level more than 1000ng/ml) with particular emphasis on cases that needed surgical intervention due to unsatisfactory results from BC therapy alone. BC was effective in 6 cases (60%). They had the serum PRL level normalized in less than one year, with BC maintenance doses between 5-15mg/day. MRI revealed complete or nearly total disappearance of the tumor. Discontinuation of the medicine was achieved in one patient. In the remaining 4 cases, surgery was needed for various reasons: 1) BC resistant prolactinomas in 2 cases, 2) large hematomas in one of the previous cases, 3) regrowth of tumor size, despite the nearly normalized PRL level due to bulk increase in non-secretory adenomatous portion in 1 case, and 4) intolerable side effect in 1 case. BC is effective even for giant prolactinomas in 60% of cases. But, this therapy needs surgical intervention more often than microadenomas. Surgical indications and timing should be decided on based on closer follow-up of neuroimaging and visual evaluation as well as the serum PRL level.

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To examine the importance of tonic LH secretion in ovarian follicular development, plasma concentrations of LH were lowered by administering antiserum to GnRH (GnRH AS). At 1100h on metestrus or diestrus of the 4-day estrous cycle, rats were injected with GnRH AS, and blood samples were collected at 6h intervals until 1100h on the next metestrus. Separately, rats were injected with hCG after the administration of GnRH AS to examine the effect of lowered LH on the number of ovulatory follicles. Treatment with GnRH AS on either metestrus or diestrus lowered plasma concentrations of LH, inhibin, and estradiol. In contrast, the plasma concentrations of FSH increased after the GnRH AS treatment, probably due to a decrease in plasma levels of inhibin associated with suppression of ovarian follicular development. Administration of GnRH AS on metestrus noticeably reduced the number of ovulatory follicles in spite of the high FSH secretion. Such a reduction in the number of ovulatory follicles was blocked by replacing rat LH with ovine LH. These data indicate that tonic secretion of LH plays an important role in the regulation of follicular development during the estrous cycle of the rat.

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We previously reported that the intraperitoneal and intrabursal administration of bestatin, an aminopeptidase inhibitor, enhanced follicular growth and ovulation in immature mice. In this study, we examined the effect of bestatin on follicular growth and ovulation under the conditions in which the ovarian response to gonadotropins was suppressed. Immature female mice were exposed to 72-h continuous lighting (on days 20-22 of life), or to 24-h starvation (on day 20). On days 20-22, bestatin (4 mg/ml, 100μl) or PBS (100μl) was administered ip. 4 times and pregnant mare serum gonadotropin (PMSG, 5IU) and hCG (5IU) were given on days 21 and 23, respectively. The numbers of ovulated oocytes per mice were significantly attenuated by both stresses. Bestatin significantly enhanced the numbers of ovulated oocytes under both lighting (49.5±7.0 vs. 28.0±5.5) and starvation (28.6±2.6 vs. 20.2±2.0) stresses (with vs. without bestatin, P<0.05). When follicular development was stimulated by PMSG (20IU) on day 21, the serum estradiol concentration on day 23 was also suppressed by both stresses, but enhanced by bestatin under lighting (231.3±36.0 vs. 111.0±23.0pg/ml) and starvation (151.9±8.8 vs. 90.7±15.4pg/ml) stresses (with vs. without bestatin, P<0.01). Northern blot analysis revealed that the expression of P450arom-mRNA in the ovaries induced by PMSG (20IU) was increased on day 23 by treatment with bestatin under starvation stress. These results indicated that stress inhibited the ovarian response to gonadotropins and that bestatin restored the response suppressed by stress.

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Abnormalities in the ret proto-oncogene are found in several disorders such as multiple endocrine neoplasia (MEN) 2, sporadic medullary thyroid cancer, congenital megacolon and papillary thyroid cancer. In MEN 2A or 2B, early genetic diagnosis before the development of clinical tumors is crucial for the cure of the disease. We studied mutations of ret proto-oncogene in 3 Korean families with MEN 2A and searched for new restriction sites that could be used for genetic diagnosis. By direct sequencing of exon 10 and 11 harboring 'hot' spots, heterozygous point mutation was detected at positions translating cysteine codon in all 3 families. In 2 families, mutations at codon 634 in exon 11 were found (from TGC to CGC or TAC), yielding a new CfoI or RsaI restriction site. In one family, a mutation was located at codon 618 in exon 10 (from TGC to CGC), generating a new CfoI restriction site. These new restriction sites were used in detecting 2 undiagnosed family members without clinical symptoms or signs. In one of them, thyroidectomy was performed to disclose a small medullary thyroid cancer. These results indicate that Korean MEN 2A patients have germ-line mutations in the ret protooncogene at the cysteine residues like patients of other races, and the strategy employing direct sequencing to find mutations at 'hot spot' and search for ensuing new restriction sites could be a useful approach for the molecular diagnosis of genetic diseases accompanied by mutations in restricted areas of disease genes such as MEN 2.

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Congenital hypothyroidism is caused by several mechanisms. The most common cause worldwide is iodine deficiency, but in iodine-sufficient regions thyroid dysgenesis is the most common cause of congenital hypothyroidism. In the present study we analyzed the thyroid transcription factor-1 (TTF-1) gene in patients with congenital hypothyroidism due to thyroid dysgenesis: three patients with athyrosis, five with ectopy, and one with hypoplasia. Genomic DNA was isolated from peripheral leukocytes, and the TTF-1 gene, including a 5' flanking region, two exons and one intron was amplified by polymerase chain reaction (PCR) with 4 pairs of primers. The PCR products were directly sequenced by the Dye Terminator Cycle Sequencing method. We could not find any mutations specific for the thyroid dysgenesis in the 5' flanking region, two exons and one intron in the TTF-1 gene, but two heterozygous nucleotide substitutions were detected in the intron: a G to A transition at nucleotide 469 (G469A) and a C to A transversion at nucleotide 866 (C866A). The same nucleotide changes were detected in some normal subjects. Allelic frequencies of the polymorphisms G469A and C866A were 23% and 10%, respectively. Another normal polymorphism in the 5' flanking region was a G to T transversion at nucleotide -845 from the transcription start site (G-845T). The allelic frequency of the polymorphism G-845T was 28%. We also found 12 polymorphisms in the 5' flanking region, two in the intron and one in the 3' untranslated region. These polymorphisms were detected in 100% chromosomes. These results suggest that congenital hypothyroidism associated with thyroid dysgenesis is unlikely to be caused by mutations in the TTF-1 gene in which, however, were detected normal polymorphisms in the 5' flanking region, intron and 3' untranslated region.

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Prohormone convertase 2 (PC2) is a member of a family of mammalian subtilisin-like endoproteases that are involved in the processing of prohormones, neuropeptides and many other precursor derived proteins. The expression of PC2 is restricted to neuroendocrine tissues such as pancreatic islets, the pituitary and the brain. To understand the regulation of the PC2 gene, we cloned and characterized the promoter region of the mouse PC2 gene. The transcriptional start site of the mouse PC2 gene is identical to that of the human. There is 79% identity in the sequences of the promoter regions between the mouse and human PC2 genes. The mouse PC2 gene, like the human, does not have a TATA-like motif in the region just upstream of the start of the transcription. Studies with promoter- reporter gene, chrolamphenicol acetyltransferase (CAT), constructs showed that the region from -400 to -170bp was necessary for high level expression of the mouse PC2 gene in the βTC-3 insulinoma cells.

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To determine the effect of nicotine on LH and PRL secretion, nicotine bitartrate (nicotine) dissolved in saline was administered at 1400h, just before the critical period for the preovulatory surge of LH and PRL secretion, either intracerebroventricularly (icv) or intravenously (iv) in female rats in proestrus. Nicotine neither at a dose of 5μg nor at a dose of 10μg injected icv at 1400h caused significant changes in the surge of LH and PRL secretion. When nicotine was given iv at a dose of 100 μg, a significant decrease in LH and PRL concentrations occurred immediately, lasting for 2h. After 1700h, LH and PRL concentrations as high as that observed after 1700h in saline-injected control rats were recovered, just as if nicotine caused a transient deficit of the surge secretion of these hormones. The results indicate that nicotine does not inhibit the preovulatory surge of LH and PRL secretion by acting at the hypothalamic level accessible via the third ventricle, but inhibits it by acting at certain other site(s).

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Diarrhea is a non-specific symptom which may be associated with Addison's disease and several causes had been demonstrated in the aetiology. We describe a patient with Addison's disease who was suffering from chronic diarrhea for three months. She was diagnosed as having collagenous colitis and successfully treated with Sulphasalazine, 2g/day. Collagenous colitis is an uncommon cause of chronic diarrhea and the association of collagenous colitis with Addison's disease has not previously been described. We think that collagenous colitis may play a role in the aetiology of diarrhea in patients with Addison's disease and therefore we suggest a full colonoscopic examination in other patients with Addison's disease and diarrhea to determine the incidence of collagenous colitis in the aetiology of diarrhea.

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We evaluated the effect of GnRH on the serum hCG level in gestational trophoblastic disease (GTD). Five patients with GTD were studied. Three patients had hydatidiform mole (two complete and one partial mole) and two had choriocarcinoma. Blood samples were collected immediately before and 30, 60min after the 100μg GnRH iv injection, followed by hCG assay. Only one case of choriocarcinoma demonstrated an hCG increase after intravenous administration of GnRH (positive GnRH test). In that case, the hCG level dropped to the normal range after eight cycles of chemotherapy but the GnRH test was still positive, suggesting the existence of viable cancer cells. Since the GnRH test became negative, no increase in hCG has been observed, indicating that the patient achieved complete remission. Although a positive GnRH test is not common in GTD, GnRH test before treatment might be useful to find a positive case where the test can be repeated to determine complete remission and the time when the chemotherapy may be discontinued.

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Chicken TSH β subunit cDNA was cloned and sequenced. The cDNA encodes a putative signal peptide and a mature protein consisting of 20 and 114 amino acids, respectively. The amino acid sequence of chicken TSH β subunit is highly conserved (98.5%) between chicken and Japanese quail, whereas it has a low homology between chicken and mammals (67-69%), an amphibian (58%) and fish (40-49%). Structural characteristics of TSH β subunits of avian species are discussed in comparison with those of non-avian vertebrates.

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We report on a girl with recurrent painful thyroiditis which involved the entire right thyroid lobe, but did not spread to the left thyroid lobe. She occasionally developed pain and swelling of the right side of the neck following symptoms of common cold over several years. During these attacks, the right thyroid lobe was enlarged, tender, and very soft, while the left thyroid lobe was not remarkable. Ultrasonic examination revealed that the entire right thyroid lobe was hypoechoic, but the entire left lobe was normal echoic. Serum C-reactive protein, white blood cell count, and erythrocyte sedimentation rate were increased during the attacks. The thyroiditis responded quickly to treatment with prednisolone and/or anti-inflammatory drugs, but the thyroiditis may have subsided spontaneously since the signs and symptoms of thyroiditis had subsided with only antibiotic therapy. After resolution of the thyroiditis, the echogenicity of the right lobe returned to near normal and the inflammatory findings became negative. During the investigation, thyroid function was normal except for when the thyroiditis was first diagnosed at another hospital with a serum TSH level of 0.1mU/l. Serum thyroglobulin level was increased but returned to normal or decreased during and after attacks of thyroiditis. The mechanism(s) of the recurrent hemithyroiditis remains unknown, but the underlying disease appeared to be chronic thyroiditis since tests for anti-microsomal or anti-thyroid peroxidase antibodies were consistently positive during the course of her illness. The changes in the titers of these antibodies were not clear during and after the attacks of thyroiditis. In addition, the clinical course suggests that the present hemithyroditis can be induced by recurrent upper respiratory infection.

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Male hypogonadism has been recognized as one of the major causes of secondary osteoporosis, but most cases seem to be left undiagnosed. We report a 54-year-old case of mosaicism Klinefelter syndrome lacking typical clinical features such as tall stature or low intelligence, who was found to have marked decrease in lumbar bone mineral density (BMD: 0.686g/cm²) during treatment of diabetes mellitus. In investigation for etiologies of secondary osteoporosis, he was diagnosed as having mosaicism Klinefelter syndrome (XXY/XY/XX). Although he was infertile, he lacked typical clinical features of Klinefelter syndrome. Testosterone replacement was started, which resulted in an increase in BMD up to 0.712g/cm² two months after the initiation of therapy. The fact that BMD increased shortly after the initiation of testosterone replacement therapy in the present case supported a beneficial effect of testosterone on BMD, as recently suggested in idiopathic hypogonadotropic hypogonadism. Although the present case was diagnosed as having mosaicism Klinefelter syndrome by investigating etiologies for osteoporosis, it may be stressed that male hypogonadism, in general, should be adequately suspected in the presence of infertility and from the findings of physical examination.

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