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Volume 14(3); September 1999
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Review Articles
Adrenal Incidentaloma.
Yeon Ah Sung
J Korean Endocr Soc. 1999;14(3):433-439.   Published online January 1, 2001
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AbstractAbstract PDF
No abstract available.
G Protein Coupled Receptor Signaling: Recent Progress.
Ja Hyun Baik
J Korean Endocr Soc. 1999;14(3):440-452.   Published online January 1, 2001
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No abstract available.
Medical Treatment of Acromegaly.
Seong Yeon Kim
J Korean Endocr Soc. 1999;14(3):453-457.   Published online January 1, 2001
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No abstract available.
Original Articles
The Effect of Slow Release Lanreotide in Korean Acromegalic Ratients.
Sang Hwa Kim, In Myung Yang, Kwang Sik Seo, Eul Soon Im, Seung Joon Oh, Deog Yoon Kim, Jeong Taek Woo, Sung Woon Kim, Jin Woo Kim, Young Seol Kim, Sun Woo Kim, Young Kil Choi
J Korean Endocr Soc. 1999;14(3):458-471.   Published online January 1, 2001
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BACKGROUND
Previous studies have shown that somatostatin analogues such as octreotide are effective in suppressing GH and IGF-I levels in acromegaly. The recent availability of slow release lanreotide could avoid the inconveniences associated with either repeated subcutaneous injections or continuous infusions. We investigated the effects of the SR-lanreotide on clinical, biochemical and safety responses in five patients with acromegaly. And we investigated whether the response of the GH level to acute adrninistration of octreotide predicts the response after 12 weeks of treatment with the SR-lanreotide and whether the identification of gsp oncogene could be used as a therapeutic and prognostic clue in treatment with the SR-lanreotide. METHODS: We studied the effects of SR-lanreotide 30 mg administered intramuscularly biweekly for 12 weeks in five Korean acromegalic patients. Subjective improvements in the clinical symptoms of acromegaly and adverse reactions were recorded. During SR-lanreotide treatment, serum GH, IGF-I and IGFBP-3 concentrations were evaluated just before the next injection of the SR-lanreotide. Before the start of SR-lanreotide therapy the sensitivity of GH secretion to the octreotide was tested by measuring the effect of the acute response to 0.1 mg intravenously on plasma GH levels followed until 6 hours after administration of octreotide. Direct polymerase chain reaction sequencing of the gsp oncogene were performed. We compared the responses to SR-lanreotide in patients harboring gsp-positive and gsp-negative somatotroph adenomas. RESULTS: The treatment with SR-lanreotide for 12 weeks could suppress the GH level by more than 50% in four of five patients and normalize the IGF-I in two patients. No correlation was found between the GH level and IGF-I level at the end of the study. The IGFBP-3 level correlated with the IGF-I level in three of five patients. Although the initial GH response to octreotide tended to correlate with the IGF-I response after SR-lanreotide treatment, the results were statistically insignificant. The patients with gsp-positive tumor tended to show a better response to SR-lanreotide. During treatment, there was a reduction in the percentage of patients complaining of joint pain, fatigue, digital paresthesia, and hyperhydrosis. Changes in soft tissue swelling were documented by decreases in finger circumference. The common adverse events were abdominal discomfort, loose stool, and diarrhea. These events were decreased progressively. No patients discontinued the treatment of SR-lanreotide due to adverse events. CONCLUSION: This study showed that SR-lanreotide is effective in controlling acromegalic symptoms as well as GH and IGF-I hypersecretion. This treatment was well tolerated and more convenient for the patients. Further studies are required for clinical outcome of long-term SR-lanreotide treatment and cost-effective analysis.
SR (Slow-Replase) Lanreotide Treatment in Acromegalic Patients.
Jae Hyun Nam, Sung Kil Lim, Sun Ho Kim, Chul Woo Ahn, Song Chul Lee, Young Duk Song, Kyung Rae Kim, Hyun Chul Lee, Ki Hyun Park, Kap Bum Huh
J Korean Endocr Soc. 1999;14(3):472-482.   Published online January 1, 2001
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BACKGROUND
Several clinical studies reported the efficacy of the long-acting SRIH analog, octreotide (Octreotide, Sandoz) in the treattnent of acromegaly. Recently, another SRIH analog (BIM 23014, Ipsen Biotech) was shown to decrease plasma GH levels in acromegalic patients. The recent availability of a long-acting formulation of BIM 23014 [slow release (SR) lanreotide] could avoid repeated sc injections or continuous sc infusions. The objective of this study was to determine the tolerability and effectiveness of the slow release (SR) somatostatin analog, SR lanreotide in active acromegaly. METHOD: Between March 1998 and May 1998, 10 patients were recruited in the prospective study carried out at Yonsei University. The effects of 6 weeks of SR lanreotide, given every 14 days at a dosage of 30 mg, im, were analyzed. All the patients completed the 6-week period of therapy. RESULTS: SR lanreotide injection produced 45% suppression of area under the curve of GH levels from the basal value on oral glucose tolerance test(OGTT). GH values on OGTT were normalized (< 2ng/mL) in 30% of patients after 6 weeks, whereas insulin-like growth factor I (IGF-I) levels were normalized in 50% of patients. No correlation was found between pretreatment GH levels and GH response to SR lanreotide or between changes in GH and IGF-I during therapy, The significant differences in response to SR lanreotide were shown between the patients with residual mass and no visible mass. During treatment, there was the significant reduction in the percentage of patients complaining of joint pain, hyperhydrosis, and paresthesias. Changes in soft tissue swelling were documented by a significant decrease in the diameter of fingers. Mild diarrhea and fatigue were the most frequent side-effects (20 30%) when SR lanreotide therapy was started. However, these side effects decreased progressively. Significant changes were noted in carbohydrate tolerance. CONCLUSIONS: These data indicate that SR lanreotide at a dose of 30 mg, im, every 14 days is an effective treatment in most unselected acromegalic patients, especially in patients with no visible mass. Tolerability to SR lanreotide therapy is high. The use of a new sustained release formulation of somatostatin analog is clearly advantageous in improving patient compliance with medical treatment for acromegaly.
Diagnosis of Cushing's Disease by Inferior Petrosal Sinus Sampling (IPSS): Evidence of False Negative Results.
Seon Hwa Lee, Hyeon Jeong Jeon, Sun Hee Park, Sun Wook Kim, Do Joon Park, Kyong Soo Park, Seong Yeon Kim, Bo Youn Cho, Hong Kyu Lee
J Korean Endocr Soc. 1999;14(3):483-492.   Published online January 1, 2001
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BACKGROUND
While inferior petrosal sinus sampling (IPSS) correctly diagnoses pituitary dependent Cushings syndrome if a significant ratio of plasma ACTH between the IPS and the peripheral blood is demonstrated, little has been said about the significance of a negative ratio in Cushings disease (e.g. false negative result). We evaluated the results of IPSS with Cushings disease, and compared them with imaging findings through transsphenoidal surgery. METHODS: 29 patients with Cushings disease underwent transsphenoidal examination of the pituitary gland from 1989 to 1998 at Seoul National University Hospital were evaluated. We compared the results of IPSS and imaging findings with sellar CT or dynamic MRI. The ratio of the ACTH concentrations at the IPS and in the peripheral blood (IPS:P ratio) and the ratio of the ACTH concentrations between the IPSs (interpetrosal ratio) were calculated before and after CRH infusion. RESULTS: With IPSS the diagnosis of Cushings disease was possible in 90% (26/29), and 3 cases in whom IPSS did not show significant IPS:P ACTH ratio were confirmed to be Cushings disease through hemihypophysectomy of lesion suspected by sellar dynamic MRI and achieved remission after operation (e.g. false negative result of diagnosis for Cushings disease by IPSS). However, accurate localization of microadenoma was achieved in only 59% (17/29). Imaging study detected microadenoma in 76% (22/29) and correctly localized in 66% (19/29). Both IPSS and imaging study precisely localized the pituitary microadenoma in 10 (34.5%) cases of 29 cases and a discrepancy between two studies existed in 7 (24.1%) cases in which the imaging study correctly localized microadenoma in 6 cases and IPSS in 1 case. CONCLUSION: Only when a significant IPS:P ACTH ratio is present can Cushings disease be established by IPSS. The absence of a significant IPS:P ACTH ratio does not necessarily imply ectopic secretion of ACfH, nor does it exclude Cushings disease. The results of lateralization by IPSS do not remove the need for transsphenoidal examination of the sella turcica because false negative result can be. IPSS and radiologic study should be complementary used in diagnosis of Cushings disease and localization of microadenoma, as IPSS can be used when sellar imaging study failed to visualize the lesion and hemihypophysectomy of suspected lesion by imaging study can be considered when IPSS did not show significant ratio of ACTH.
Differential Roles of Transcriptional Coactivators: CBP and CIITA on GAS (Interferon-r Activated Site) - Mediated Transcription in Thyroid Cells.
Eun Shin Park, Ho Kim, Soon Hee You, Soo Jung Park, Hyun Jin Kim, Soo Heung Chae, Do Hee Kim, Hee Jeong Han, O Yu Kwon, Young Kun Kim, Minbo Shong, Heung Kyu Ro
J Korean Endocr Soc. 1999;14(3):493-504.   Published online January 1, 2001
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BACKGROUND
In the previous studies, we identified that the interferon-gamma activated sequence (GAS) in the 5-flanking region of rat ICAM-1 gene is major element for interferon-y-inducible expression of the gene in rat thyroid cells, FRTL-5. We here, investigated the role of transcriptional coactivators, CBP (CREB binding protein) and CIITA (class II transactivator) in the modulation of the activity of GAS which could interacts with signal transducers and activators of transcription-1 and 3 (STAT1 and STAT3). METHODS: The expression of CBP RNA and protein were quantitated in FRTL-5 after stimulation with interferon-y (IFN-gamma), thyroid stimulating hormone (TSH), forskolin and methimazole. Direct association of CBP with STAT were analyzed by irnmunoprecipitation. The transcriptional roles of CBP and CIITA in the regulation of GAS were assessed by the cotransfection with their expression vectors with reporters; 5-deletion constructs of rat ICAM-1 promoter or 8xGAS-luc constructs, into FRTL-5 thyroid cells. RESULTS: The level of CBP RNA and protein were not changed by the treatment with TSH, IFN-y, forskolin and methimazole in FRTL-5, FRT and BRL liver cells. The CBP could be directly associated with STAT1. Furthernmore, the overexpression of CBP significantly increases the both promoter activities; rat ICAM-1 gene promoter which has GAS element and 8xGAS-luc cassette constructs. However the cotransfection of CI1TA decreased the constitutive and CBP-mediated transactivation of rat ICAM-1 promoter and SxGAS-luc cassette constructs. CONCLUSION: We identified that the two transcriptional coactivators; CBP and CIITA has differential roles in the regulation of transcriptional activity of GAS drived promoter. CBP increases the GAS activity through the direct binding with STATl, but CIITA inhibited the CBP-mediated transactivation of GAS activity.
Clinical Features Associated with an Increased Risk of Malignancy in Indeterminate Group by Find Needle Aspiration of Thyroid Nodules.
Seon Hwa Lee, Sun Wook Kim, Do Joon Park, Won Bae Kim, Seong Yeon Kim, Bo Youn Cho, Hong Kyu Lee, Chang Soon Koh
J Korean Endocr Soc. 1999;14(3):505-513.   Published online January 1, 2001
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BACKGROUND
The recommended management of patients who have cytologic diagnosis of indeterminate group by fine fine needle aspiration (FNA) in thyroid nodules is controversial. Our objective was to identify the incidence of malignancy through thyroidectomy in indeterminate group and clinical features associated with an increased risk of malignancy that might guide future practice. METHODS: We retrospectively reviewed the medical records of 222 patients who had cytologic diagnosis of indeterminate group by FNA in thyroid nodules at Seoul National University Hospital from Jan. 1990 to Aug. 1998. Patients characteristics and clinical features were compared between benign and malignant nodules classified as pathologic findings through thyroidectomy. RESULTS: The frequency of indeterminate group was 6% (222/3981) among patients underwent thyroid FNA. The frequency of malignant nodules was 47.4% (64/135) among 135 patients underwent thyroidectomy. Among clinical features, rapid increase in size (7.0% vs 20.3%, p=0.024), local symptoms such as dysphagia, hoarseness, pain (1.4% vs 15.6%, p=0.003), fixation (4.4% vs 35.7%, p<0.001), hard consistency(15.7% vs 59.7%, p<0.001) and irregular surface(6.3% vs 25.6%, p=0.001) were significantly more common in malignant nodules than in benign nodules. However, clinical features such as mean age of patients, male sex, presence of past history of benign thyroid disease, family history of benign thyroid disease, solitary nodule, presence of cervical lymph nodes, mean size of nodules and cold nodules by thyroid scan in malignant nodules were not significantly different from that in benign nodules. Among clinical features that were significantly more common in malignant nodules, fixation(p=0.042) and presence of local symptoms (p=0.043) were significantly independent risk factors predictive of malignancy. CONCLUSION: Its better to recommend thyroidectomy in patients with clinical features such as presence of local symptoms, fixed nodule and hard nodule in indeterminate group, in otherwise to decide treatment with repeated FNAs when clinical features such as patients symptoms and physical examinations of nodule change through regular follow-up.
Clinical Significance of Human Sodium Iodide Symporter mRNA Expressions in Primary and Metastatic Papillary Thyroid Carcinoma.
Seong Jin Lee, Hyun Joo Park, Eun Ju Lee, Ha Young Kim, Jin Kyu Koh, Ki Young Park, Sung Bae Kim, Gyung Yup Gong, Suk Joon Hong, Il Min Ahn, Sang Hee Kim
J Korean Endocr Soc. 1999;14(3):514-519.   Published online January 1, 2001
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BACKGROUND
The iodide transport into thyroid cells is an essential step in the biosynthesis of thyroid hormones. The sodium iodide symporter (NIS) which is responsible for iodide transport was cloned recently and identified as a plasma membrane glycoprotein. Recent report suggested the absence of human NIS (hNIS) mRNA expression of papillary carcinoma in thyroid indicates absence of response on radioiodine therapy for distant metastasis. To understand the change of hNIS expression at the stage of metastasis in papillary thyroid carcinomas, we evaluated the expression levels of hNIS mRNA in primary and lymph node metastatic papillary carcinoma tissues. METHODS: Seven pairs of primary and lymph node metastatic tissues were included in this study. The level of hNIS mRNA in lymph node metastatic tissues and primary tissues were evaluated by reverse transcriptase-polymerase chain reaction (RT-PCR). The level of GAPDH mRNA was used as internal control. RESULTS: Two among 6 lymph node metastatic tissues did not show hNIS mRNA even with significant hNIS expressions in papillary carcinoma tissues in thyroid. The levels of hNIS expression of remaining 4 lymph node metastatic tissues were lower than those of corresponding primary tissues. Interestingly, one case showed no hNIS expression in primary tissue, but significant hNIS expression in lymph node metastatic tissue. There was no correlation in hNIS mRNA expression between primary and lymph node metastatic tissues. CONCLUSION: No correlation was found in hNIS mRNA expression between primary and lymph node metastatic tissues, suggesting the measurements of hNIS mRNA level in primary tissues may not predict therapeutic response to radioactive iodine.
FDG-PET as a Predictor of Recurrence with 131I Scan Negative Differentiated Thyroid Cancer: An Evaluation Compared with Pathologic Findings.
Eunju Lee, Sung Jin Lee, Cheol Ryoung Lee, Ha Young Kim, Hun Ho Song, Young So, Jin Sook Ryu, Dae Hyuk Moon, Suk Joon Hong, June Kkey Chung, Il Min Ahn
J Korean Endocr Soc. 1999;14(3):520-530.   Published online January 1, 2001
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BACKGROUND
FDG-PET has been suggested to have a supplementary role in localizing recurred sites of differentiated thyroid carcinoma. This study was performed to show whether FDG-PET is feasible as an alternative diagnostic modality for patients with I-131 scan negative thyroid carcinoma by verification of post-surgical pathology findings. METHODS: Eighteen patients of papillary thyroid carcinoma (M:F=4:14, age 41+/-16 year) who had total thyroidectomy and I-131 ablation therapy were included. All patients showed negative I-131 scan on therapeutic dose but they were suspected as disease recurrence because of elevated serum Tg or anti-Tg Ab during follow-up periods. FDG-PET was performed, and then cervical lymph node dissection on either side or both sides of the neck was done according to FDG-PET results. RESULTS: A total of 77 cervical lymph node groups were dissected in 18 patients; internal jugular chain 49, spinal accessory 9, jugulodigastric 5, anterior jugular 4, paratracheal 3, supraclavicular 2, and others 5. Forty eight lymph node groups revealed metastatic papillary carcinoma on pathology and their largest diameter ranged from 0.4 to 7.0cm (1.2+/-0.7cm). All patients had at least one malignant lymph node group. FDG-PET detected 37 among 48 malignant lymph nodes (sensitivity 77%), and their count ratio ranged 1.7-31.1 (6.1+/-6.3). Among the 30 malignant lymph nodes less than 1cm, FDG-PET detected 20 lymph nodes. Of the 29 lymph node groups without malignant cells, FDG-PET was also negative in 24 groups (specificity S3%). Positive predictive value of FDG-PET on I-131 scan negative differentiated thyroid carcinoma was 88%; negative predictive value was 69%. CONCLUSION: FDG-PET has been confirmed as a valuable diagnostic modality to detect cervical lymph nodes of differentiated thyroid carcinoma who are suspicious for recurrence but with negative I-131 scan, by pathologic findings.
The Growth Hormone (GH) - Binding Protein in Obesity with Varying Glucose Tolerance: Relationship to Body fat Distribution Sex Hormones, Insulin and GH-Insulin-Like Growth Factor (IGF)-1 Axis.
Su Youn Nam, Kyung Wook Kim, Sang Won Ji, Se Jung Yoon, Kyung Rae Kim, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh
J Korean Endocr Soc. 1999;14(3):531-540.   Published online January 1, 2001
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BACKGROUND
As GHBP is believed to be derived from proteolytic cleavage of the extracellular domain of the GH receptor and may be regarded as an intrinsic part of the GH-IGF-1 axis, an effect of body composition on circulating GHBP levels may be expected. We investigated GHBP variations in obesity with varying glucose tolerance and its relationship to body fat distribution, sex hormones, insulin secretion, and the GH-IGF-1 axis. METHODS: Bioelectrical impedence for measurement of total body fat and computed tomography for visceral fat and subcutaneous fat at umbilicus level were performed in 69 obese Koreans and 21 lean Koreans. Insulin secretion in response to an oral glucose tolerance test (OGTT) and a GH stimulation test by L-dopa, growth hormone-binding protein (GHBP), insulin-like growth factor (IGF)-1 and sex hormones (estrone, estradiol, total and free testosterone) were measured. RESULTS: Obese type 2 DM group had the highest GHBP levels and the most visceral fat amount. GHBP levels were most strongly correlated with the ratio of visceral fat area to body weight (VWR) above other parameters (r=0.725, p<0.001). Insulin- and free fatty acid-area under the curve (AUC) during OGTT and IGF-1 level were also positively correlated with GHBP levels (r=0.474, p<0.005; r=0.572, p<0.005; r=0.453, p<0.005). GH-AUC to L-dopa stimulation test was negatively correlated with GHBP levels (r=0.432, p<0.005). The GHBP level was slightly higher in females than in male in the same glucose tolerance category. In males, total and free testosterone levels were negatively correlated with GHBP levels (r=-0.516, p<0.001;r=-0.653, p<0.001). Stepwise multiple linear regression analysis showed that VWR, FFA-and insulin-AUC significantly contributed to the variability of GHBP (r=0.58). CONCLUSION: We demonstrated that 1) visceral fat amount was mainly determined GHBP levels in obese subjects with varying glucose tolerance; 2) hyperglycemia per se did not influence GHBP level, whereas insulin and FFA could play a role in regulation of GHBP level. 3) The constant concentration of IGF-1 despite GH hyposecretion suggests that increased GHBP level retlect GHBP hypersensitivity in order to compensate for decreased GH secretion in obesity; 5) the lower level of GHBP in males might be explained at least in part by a suppressive effect of androgen.
The Effect of Body Fat Disribution on Glucose, Lipid Metabolism and Grewth Hormone Secretion in Obesity.
Ae Jung Huh, Byeong Kee Choi, Dae Ho Chung, Kyung Wook Kim, Su Youn Nam, Kyung Rae Kim, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh
J Korean Endocr Soc. 1999;14(3):541-552.   Published online January 1, 2001
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BACKGROUND
Body fat distribution, rather than the level of obesity per se, appears to be a strong predictor of abnormalities in metabolic complication. Visceral fat accumulation is significantly correlated with glucose intolerance and constitutes as an independent risk factor for the diabetes mellitus. METHODS: We investigated the impact of body fat distribution on the glucose, lipid metabolism and growth hormone secretion in obese subjects with varying glucose tolerance and lean controls matched with sex and age. 69 obese Koreans (34 men, 35 women; 43.8 yrs) and 21 lean Koreans (10 men, 11 women; 40.8 yrs) were recruited. Anthropometric measurement and impedence for measurement of total body fat, and computed tomography for visceral and subcutaneous fat area at umbilicus level were performed. All subjects underwent a standard oral glucose tolerance test and GH stimulation test by L-dopa. RESULTS: The results are summarized as follows. 1. Obese patients had greater ideal body weight (%, IBW) and lean body mass (LBM) than lean controls. But no significant differences were found in IBW and LBM between 3 obese groups. 2. The 25 obese NIDDM had the highest FFA-AUC during OGTI and the lowest GH-AUC to L-Dopa stimulation test. The insulin-AUC during OGTT was the highest in 24 obese subjects with normal glucose tolerance. 3. All male groups have VSR of more than 0.4, which has been designated visceral fat obesity. In contrast all female groups have VSR of lesser than 0.4 but obese DM subjects have the highest VSR. Visceral fat area per body weight ratio(VWR) showed increasing tendency in obese, IGT, and DM group. 4. Waist circumference and VWR showed strong correlation with metabolic parameters among anthropometric parameters. They were positively correlated with FFA-AUC during OGTT and negatively correlated with GH-AUC to L-dopa stimulation. CONCLUSION: Visceral fat accumulation are associated with insulin resistance, dyslipidemia and impairment of growth hormone secretion via increase of free fatty acid. The simple waist circumference may provide a more practical indicator that correlated with aMominal fat distribution and metabolic complications associated with obesity.
Association of Estrogen Receptor Genotypes with Serum Lipids and Responsiveness of Serum Lipids to Hormonal Replacement Therapy in Korean Postmenopausal Women.
So Ra Park, Jae Eun Park, Chung Kyu Hwang, Phil Ho Jung, Chang Hoon Yim, Ho Yeon Chung, Ki Ok Han, Hyun Ku Yoon, Hak Chul Jang, In Kwon Han
J Korean Endocr Soc. 1999;14(3):553-561.   Published online January 1, 2001
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BACKGROUND
Several biologically plausible mechanisms have been proposed for estrogen-mediated caridoprotection, including estrogen-assocaited changes in lipid metabolism and endothelial function of vessel walls. These effects are thought to be mediated via estrogen receptor (ER). Relationships between ER polymorphisms and serum lipid levels were not investigated enoughly. METHODS: Three restriction fragment length polymorphisms (RFLPs) at the ER gene locus, represented as B-variant, PvuII and XbaI, and their relationship to serum lipid levels were examined in 318 postmenopausal women. Their mean age was 54.5+/-6.5 years (mean+SD). An association between ER genotypes and changes in lipid levels after 1 year of estrogen replacement therapy was also investigated in follow-up 251 women. RESULTS: The B-variant was not found in Korean women. The distribution of the PvuII and XbaI polymorphisms was as follows: PP 109 (34%), Pp 166 (52%), pp 43 (14%), and XX 204 (64%), Xx 95 (30%), xx 19 (6%). Significant relationship was found between genotypes and changes in serum total cholesterol levels after lyr estrogen replacement therapy. There was no significant relationship between ER genotypes and changes in HDL cholesterol, LDL cholesterol and triglyceride levels after estrogen therapy. CONCLUSION: These data indicate that these polymorphisms are possible predictor on lipid response to estrogen replacement therapy.
Case Report
A Case of Normal Full Term Delivery after Afrenalectomy for Cushing's Syndrome in a Pregnant Patient.
Sung Sik Yang, Yong Seong Kim, Yong Bum Cho, Young Wan Kim, Seong Bin Hong, Yeo Joo Kim, Mi Rim Kim, Moon Suk Nam, Sei Joong Kim, Suk Hwan Shin, Byoung Ick Lee
J Korean Endocr Soc. 1999;14(3):562-567.   Published online January 1, 2001
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AbstractAbstract PDF
We experienced a case of Cushings syndrome due to adrenal adenoma associated with pregnancy 23th week in a 26 year old female patient. On physical findings, hypertension, moon face, hirsutism, abdominal striae and buffalo hump were noticed. Laboratory findings showed increased serum cortisol, increased 24 hours-urinary cortisol, and decreased plasma ACTH. Abdominal MRI demonstrated 4 cm sized in long diameter, well marginated, and ovoid-shape right adrenal mass. Unilateral total adrenalectomy was performed at the 23th week of pregnancy and the mass it was confirmed to benign adrenal adenoma. Steroid replacement therapy was continued with 7.5 mg prednisolone during remaining pregnancy. At the 38th week of pregnancy, labor was developed and she delivered healthy female weighing 2.5 kg through normal vaginal delivery without complication. Pregnancy rarely occurs in patients with Cushings syndrome. We think she was the first case in Korea who had normal 38th week transvaginal delivery after adrenalectomy in adrenal Cushings syndrome during pregnancy.
Original Article
Clinical and Hormonal Analysis of Five New Cases and Eight Previously Reported Cases of Isolated ACTCH Deficiency in Korea.
Eun Young Oh, Byoung Joon Kim, Yun Jae Chung, Dong Joon Kim, Jong Ryul Hahm, Jae Hoon Chung, Yong Ki Min, Myung Shik Lee, Moon Kyu Lee, Kwang Won Kim
J Korean Endocr Soc. 1999;14(3):568-577.   Published online January 1, 2001
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Isolated ACTH deficiency is an uncommon disorder, which is defined by low cortisol production with low or normal plasma ACTH levels and no other pituitary abnormalities. We report five new cases of this disorder, and summarize the clinical and hormonal features of 8 previously reported cases in Korea plus 5 new cases. 1) The clinical manifestations of isolated ACTH deficiency are variable, non-specific and similar to those seen in adrenocortical insufficiency of any cause, the age of patients ranged from 21 to 66 years old with an average age of 46 years, and the male to female ratio was 10:3. 2) Hyponatremia and hypoglycemia were commmon laboratory findings, so the presence of unexplained hyponatremia or hypoglycemia should always warrant consideration of the diagnosis of isolated ACTH deficiency. 3) 3 of 13 patients accompanied by empty sella suggesting selective destruction of pituitary ACTH producing cells. 4) ACTH response to exogenous CRH or vasopressin was not elicited in all tested cases, suggesting pituitary disorders. 5) Most patients showed dramatic response with oral predinisone. In conclusion, when there are unexplained general weakness, fatigue, weight loss, nausea, vomiting, hypoglycemia, or hyponatremia, isolated ACTH deficiency should be excluded. Immunologic and pathologic studies, and hormonal evolution with glucocorticoid treatment are needed to understand the pathogenesis of isolated ACTH deficiency.

Endocrinol Metab : Endocrinology and Metabolism