Endocrinology and Metabolism

Case Reports

A Case of Diabetes Insipidus with Langerhans Cell Histiocytosis in Adult.: Jae Hoon Chung, Kwang Won Kim, Kyu Jeung Ahn, Yong Ki Min, Myung Shik Lee, Moon Kyu Lee, Eun Mi Koh, Choon Kwan Kim, Mann Pyo Jung; J Korean Endocr Soc. 1996;11(3):330-335. Published online November 7, 2019

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Abstract PDF: In Langerhans cell histiocytosis, diabetes insipidus is the most common endocrinologic complication. We experienced a case of Langerhans cell histiocytosis, involving pituitary stalk and lung. The patient was a 43 year old male with complaint of polyuria and polydipsia. The water deprivation test was carried out to confirm the diagnosis of diabetes insipidus. We found multiple small cysts and nodules in HRCT of lung, and diagnosed Langerhans cell histiocytosis by transbronchial lung biopsy, The patient was managed conservatively with DDAVP nasal spray. The polyuria,polydipsia was relieved completely. After that, we follow up and observe closely the patients lung and pituitary lesion.

Two cases of 111Indium Pentetreotide Scan for the Pre- and Post-Operative Evaluation of Localization and Metastasis in Medullary Thyroid Carcinoma.: Jae Hoon Chung, Kwang Won Kim, Kyu Jeung Ahn, Yong Ki Min, Myung Shik Lee, Moon Kyu Lee, Kyoung Ah Kim, Yeun Sun Kim, Eun Mi Koh; J Korean Endocr Soc. 1996;11(1):85-92. Published online November 7, 2019

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Abstract PDF: Medullary carcinoma of the thyroid gland(MTC) constitutes approximatesly 3% to 10% of all malignant thyroid tumors. It appears in both familial and sporadic forms. Metastases are frequently present at diagnosis and are resistant to chemotherapy and radiotherapy. Surgical resection of the primary tumor and the metastases is the mainstay of treatment. Although MTC can be detected by elevated serum calcitonin, localization of residual or metastatic foci may be difficult. Many scintigraphic methods have been used for identification of the residual tumor or metastasis. However, most of them have either low sensitivity or low specificity. MTC frequently secretes somatostatin and may express somatostatin receptors. Recently, somatostatin-receptor imaging has been known to be useful for the detection of residual and recurrent medullary thyroid carcinoma. A 25 year-old woman who was dignosed as medullary carcinoma by biopsy of thyroid mass is presented. Thirteen years ago, she underwent left thyroidectomy due to thyroid cancer(MTC). Laboratory tests revealed an increase in the levels in serum CEA(CEA=557.6 ng/ml) and calcitonin(calcitonin= 720 pg/ml). The second patient, a 30 year-old female, complained of a palpable mass in the left anterior neck. Ten years ago, she underwent a right lobectomy of thyroid gland due to adenomatous goiter. Laboratory tests revealed an increase in the levels in serum CEA(CEA=617 ng/ml) and Calcitonin (Calcitonin=2,300 pg/ml). In both cases, pre- and postoperative In-111 pentetreotide scintigraphy were done and compared with "'I scintigraphy. In-111 pentetreotide scan may be useful for the localization of residual or metastatic medullary thyroid carcinoma. Further study is warranted to define the sensitivity and specificity of the technique.

Original Article

Spontaneous Recovery from Hypothyroidism in Autommune Thyroiditis.: Bo Youn Cho, Jae Hoon Chung, Kwang Won Kim, Kyu Jeung Ahn, Yong Ki Min, Myung Shik Lee, Moon Kyu Lee; J Korean Endocr Soc. 1996;11(1):30-40. Published online November 7, 2019

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Abstract PDF: Background
A lifelong thyroxine therapy is indicated in all patients who have hypothyroidism as a result of autoimmune thyroiditis. However, it has been reported that some hypothyroid patients with autoimmune thyroiditis have spontaneous remission with restriction of iodine intake instead of thyroxine therapy. The purpose of study was to investigate how many hypothyroid patients with autoimmune thyroiditis can recover from hypothyroidism with restriction of iodine intake instead of thyroxine therapy and which factors predict recovery from hypothyroidism. Methods: We studied 64 patients with autoimmune thyroiditis(goitrous autoimmune thyroiditis 56, atrophic autoimmune thyroiditis 8). Thyroxine therapy was discontinued in patients with goitrous autoimmune thyroiditis on the way(group 1, n=32) or from the beginning(group 2, n=24) and atrophic autoimmune thyroiditis on the way(group 3, n-8). All patients were asked to avoid iodine-rich foods and thyroid function was monitored every one to two months for up to 35 months. Serum T3, T4, TSH concentrations, antithyroglobulin and antimicrosomal antibodies were measured by radioimmunoassay(RIA). TSH binding inhibitor immunoglobulin(TBII) was measured in serum using radioreceptor assay. Two hundred micrograms of thyrotropin releasing hormone (TRH) were given as intravenous bolus and TSH levels were measured in blood samples taken at 0, 30, and 60 minutes. All values were expressed as mean+-SEM. Statistical analysis was done with paired or non-paired t-test, ANOVA, and the Chi-square test. Statistical significance was defined as p-value below 0.05. Results: Thirteen(40.6%) of 32 patients in group 1 remained euthyroid after 12-35 months of discontinuation of thyroxine therapy. The other 19(59.4%) patients in group 1 had recurrences of hypothyroidism within 3 months after discontinuation of thyroxine therapy. In 11(45.8%) out of 24 patients in group 2, serum TSH concentrations declined below 5 mU/L within 3 months without thyroxine therapy. The other 13(54.2%) patients in group 2 remained hypothyroid till 2-16 months and the thyroxine was given. In contrast, all 8 patients in group 3 had recurrences of hypothy- roidism within 3 months after stopping thyroxine therapy. When we compared the recovered patients of goitrous autoimmune thyroiditis with the non-recovered patients of goitrous autoimmune thyroiditis, regardless of thyroxine therapy from the beginning, age at onset of disease of the 24 recovered patients was significantly younger than the 32 non-recovered patients(30.1+2.0 years vs. 40.2+ 2.4 years; p=0.004). Concl#usion: These findings suggest that 42.9% of hypothyroid patients with goitrous autoim- mune thyroiditis remain or become spontaneously euthyroid with restriction of iodine intake instead of thyroxine therapy. Young age may be a predicting factor of recovery from hypothyroidism in goitrous autoimmune thyroiditis.

Case Reports

Adrenal gland
Acromegaly with Normal Insulin-Like Growth Factor-1 Levels and Congestive Heart Failure as the First Clinical Manifestation: Hyae Min Lee, Sun Hee Lee, In-Ho Yang, In Kyoung Hwang, You Cheol Hwang, Kyu Jeung Ahn, Ho Yeon Chung, Hui-Jeong Hwang, In-Kyung Jeong; Endocrinol Metab. 2015;30(3):395-401. Published online December 9, 2014; DOI: https://doi.org/10.3803/EnM.2015.30.3.395

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Abstract PDF PubReader

The leading cause of morbidity and mortality in patients with acromegaly is cardiovascular complications. Myocardial exposure to excessive growth hormone can cause ventricular hypertrophy, hypertension, arrhythmia, and diastolic dysfunction. However, congestive heart failure as a result of systolic dysfunction is observed only rarely in patients with acromegaly. Most cases of acromegaly exhibit high levels of serum insulin-like growth factor-1 (IGF-1). Acromegaly with normal IGF-1 levels is rare and difficult to diagnose. Here, we report a rare case of an acromegalic patient whose first clinical manifestation was severe congestive heart failure, despite normal IGF-1 levels. We diagnosed acromegaly using a glucose-loading growth hormone suppression test. Cardiac function and myocardial hypertrophy improved 6 months after transsphenoidal resection of a pituitary adenoma.

Citations

Citations to this article as recorded by

Risk of Neurodegenerative Diseases in Patients With Acromegaly
Sangmo Hong, Kyungdo Han, Kyung-Soo Kim, Cheol-Young Park
Neurology.2022;[Epub] CrossRef
Levels of Serum IGF-1, HCY, and Plasma BNP in Patients with Chronic Congestive Heart Failure and Their Relationship with Cardiac Function and Short-Term Prognosis
Zhengyi Hu, Leifang Mao, Ling Wang, Weiguo Li
Evidence-Based Complementary and Alternative Medicine.2022; 2022: 1. CrossRef
Neurofibromatosis Type 1 with Concurrent Multiple Endocrine Disorders: Adenomatous Goiter, Primary Hyperparathyroidism, and Acromegaly
Shigemitsu Yasuda, Ikuo Inoue, Akira Shimada
Internal Medicine.2021; 60(15): 2451. CrossRef
Metformin stimulates IGFBP-2 gene expression through PPARalpha in diabetic states
Hye Suk Kang, Ho-Chan Cho, Jae-Ho Lee, Goo Taeg Oh, Seung-Hoi Koo, Byung-Hyun Park, In-Kyu Lee, Hueng-Sik Choi, Dae-Kyu Song, Seung-Soon Im
Scientific Reports.2016;[Epub] CrossRef

A Case of Fulminant Type 1 Diabetes Mellitus with Human Leukocyte Antigen DR4-DQ4.: Ye Ri So, Ja Won Koo, Young Hak Cho, You Cheol Hwang, Kyu Jeung Ahn, Ho Yeon Chung, In Kyung Jeong; Endocrinol Metab. 2012;27(4):314-317. Published online December 20, 2012; DOI: https://doi.org/10.3803/EnM.2012.27.4.314

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Abstract PDF: The clinical characteristics of fulminant type 1 diabetes are abrupt onset of disease, very short (<1 week) duration of diabetic symptoms, ketoacidosis at diagnosis, negativity for islet-related autoantibodies, virtually no C-peptide secretion (fasting plasma C-peptide <0.3 ng/mL), a near normal hemoglobin A1c (HbA1c) level and an elevated serum pancreatic enzyme level. The pathogenesis has not yet been clarified, however the involvement of both genetic background and viral infections has been suggested. We reported a case of fulminant type 1 diabetes. A 37-year-old woman was admitted with stuporous consciousness to our hospital. Four days prior to the admission, she was hospitalized with the diagnosis of acute pancreatitis in another hospital, and at that time her glucose level was 79 mg/dL. Three days after the hospitalization, her state of consciousness became stuporous and she was transferred to our hospital. The laboratory results were as follows: pH 6.94, glucose 1,602 mg/dL, and HbA1c 5.5%. She was negative for islet-related autoantibodies and viral antibodies. HLA haplotypes were DRB1*04:05/*04:06, DQB1*03:02/*04:01 which might be a considerable risk allele for fulminant type 1 diabetes. She was diagnosed with fulminant type 1 diabetes, and has been treated with multiple component insulin regimens.

A Case of Primary Adrenal Insufficiency in a Patient with Acquired Immunodeficiency Syndrome.: Jae Ho Choi, Suk Chon, Yu Chul Hwang, Jun Seong Son, Seung Joon Oh, Kyu Jeung Ahn, Ho Yeon Chung, Jeong Taek Woo, Sung Woon Kim, Jin Woo Kim, Young Seol Kim, In Kyung Jeong; Endocrinol Metab. 2011;26(3):253-257. Published online September 1, 2011; DOI: https://doi.org/10.3803/EnM.2011.26.3.253

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Abstract PDF

The adrenal gland is the most commonly involved endocrine organ in patients infected with the human immunodeficiency virus (HIV). Adrenal function abnormality is more common in HIV patients than in the general population. It is important to recognize the condition of adrenal insufficiency, as this adrenal disorder may prove fatal if left untreated. Herein, we report a case of primary adrenal insufficiency in a 37-year-old male patient with acquired immunodeficiency syndrome. The patient complained of fever, general weakness, and fatigue. Impaired adrenal function was noted in the rapid ACTH stimulation test. After steroid supplementation, the patient's symptoms were improved. Therefore, HIV care physicians should ascertain adrenal dysfunction in HIV patients when they complain of fever and general weakness.

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A Case Report of Adrenal Insufficiency Treated with Korean Medicine
Young-ji Kim, Jung-yeon Kwon, Ho-yeon Go, Kyung-hwan Kong
The Journal of Internal Korean Medicine.2017; 38(5): 583. CrossRef

A Case of Adrenocortical Carcinoma Secreting Cortisol, Androgen and Aldosterone.: Jae Ho Choi, Ye Ri So, Yu Chul Hwang, In Kyung Jeong, Kyu Jeung Ahn, Ho Yeon Chung, Seung Ae Yang; Endocrinol Metab. 2011;26(3):239-242. Published online September 1, 2011; DOI: https://doi.org/10.3803/EnM.2011.26.3.239

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Abstract PDF

Primary adrenocortical carcinoma is a rare tumor, and is characterized by a peri-tumor mass effect and hormone excess signs. Adrenocortical carcinoma most commonly secretes cortisol, but tumors that secrete other adrenal hormones (aldosterone, androgen) are rare. Herein, we report the case of a 70-year-old woman with cortisol, androgen, and aldosterone-secreting adrenal carcinoma. The patient complained of generalized weakness, moon face, and central obesity. On laboratory examination, hypokalemia and metabolic alkalosis was detected. On the hormone test, cortisol, DHEA-S, and aldosterone were all increased. Abdominal CT showed a large right adrenal mass. She underwent right adrenalectomy and the histology revealed the presence of an adrenocortical carcinoma. After adrenalectomy, the patient was treated with hydrocortisone and mitotane.

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A Case of Adrenocortical Carcinoma Secreting Cortisol and Aldosterone
Jiyoon Ha, Min Kyung Kim, Yoon Jin Cha, Seung Kyu Kim, Gi Young Yun, Kwangwon Rhee, Joon Seong Park, Eun-Suk Cho, Chul Woo Ahn, Jong Suk Park
Yeungnam University Journal of Medicine.2012; 29(2): 132. CrossRef

A Case of Type I Osteogenesis Imperfecta Differentially Diagnosed as a Cause of a Spinal Compression Fracture.: Sang Youl Rhee, Soo Young Moon, Suk Chon, In Kyung Jeong, Seungjoon Oh, Kyu Jeung Ahn, Ho Yeon Chung, Jeong Taek Woo, Sung Woon Kim, Young Seol Kim, Jin Woo Kim; J Korean Endocr Soc. 2007;22(6):446-452. Published online December 1, 2007; DOI: https://doi.org/10.3803/jkes.2007.22.6.446

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Abstract PDF: Osteogenesis imperfecta (OI) is a genetic disease that is caused by a synthetic anomaly of type I collagen. It is characterized by such features as low bone density, multiple fractures, bone deformities and chronic bone pain. According to the hereditary pattern and degree of phenotypical expression, it also has various extraskeletal manifestations such as blue sclera, hearing deformities and dentinogenesis imperfecta. Recently, an expanded seven subgroup classification of OI has been suggested by means of its clinical severity and mutational characteristics. However, most of the OI cases reported in Korea have been classified as type II or III that can be diagnosed easily and present with severe clinical manifestations. Only rare type I OI cases have been currently reported in Korea. Herein, we report a case of type I OI that was differentially diagnosed as a cause of a spinal compression fracture.

A Case of Osteomalacia Caused by Severe Vitamin D Deficiency.: Seok Yeon Kim, Sang Youl Rhee, Soo Young Moon, Suk Chon, In Kyung Jeong, Seungjoon Oh, Kyu Jeung Ahn, Deog Yoon Kim, Ho Yeon Chung, Sung Woon Kim, Jin Woo Kim, Young Seol Kim, Jeong Taek Woo; J Korean Endocr Soc. 2007;22(1):55-61. Published online February 1, 2007; DOI: https://doi.org/10.3803/jkes.2007.22.1.55

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Abstract PDF

Vitamin D is a kind of hormone that can be synthesized in the skin or it is supplied in the diet it is involved in a central role in bone and calcium homeostasis in vivo. Metabolic bone diseases such as rickets in children and osteomalacia in adults can also be caused by deficiency or metabolic defects in the vitamin D hormone related system. However, there has been a lack of generalized epidemiologic studies about the vitamin D deficiency status in Koreans. We recently experienced a case of osteomalacia caused by severe vitamin D deficiency. These kinds of case have not been reported elsewhere in Korea for the last twenty years.

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A Case of Nutritional Osteomalacia in Young Adult Male
Choong-Kyun Noh, Min-Jeong Lee, Bu Kyung Kim, Yoon-Sok Chung
Journal of Bone Metabolism.2013; 20(1): 51. CrossRef

A Case of Islets Cell Hyperplasia Diagnosed by Selective Arterial Calcium Stimulation Test.: Jin Hoon Youn, Kyu Jeung Ahn, Yeong Shil Joo, Byoung Joon Kim, Sung Hoon Kim, Kye Hyoung Kwon, Myung Ah Jung, Jin Soo Yang, In Su Jung, Chung Hyeon Kim, Sung Bum Cho, Tae Seok Lee, Eun Kyung Kim; J Korean Endocr Soc. 2001;16(4-5):514-519. Published online October 1, 2001

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Abstract PDF: We report a 40-year-old female patient with clinical findings suggestive of insulinoma. Although imaging studies did not reveal any tumors in the pancreas, a selective arterial calcium stimulation test(SACI), procedurally simpler and more effective than transhepatic pancreatic venous sampling, was performed. And then near total pancreatectomy was carried out because the possibility of small insulinoma could not be completely excluded. Grossly, the surgically removed pancreas did not reveal any tumors. However, the pancreas exhibited islets cell hyperplasia. To our knowledge, this is the first reported authentic case in a Korean adult of islet-cell hyperplasia diagnosed by selective arterial calcium stimulation test.

A Case of Down's Syndrome with Graves' Disease.: Kap Bum Huh, Kyoung Ah Kim, Jae Hoon Chung, Yeun Sun Kim, Kyu Jeung Ahn, Eun Mi Koh, Young Ki Min, Myung Shik Lee, Moon Kyu Lee, Kwang Won Kim, Hyun Kyun Ki; J Korean Endocr Soc. 1997;12(1):61-67. Published online January 1, 2001

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Abstract PDF: Down's syndrome (trisomy 21) has been frequently associated with thyroid disease, mainly subclinical hypothyroidism (12.5-32.5%). The occurrence of Downs syndrome in conjunction with hyperthyroidism is rare (0.6-2.5%). The mechanism that Down's syndrome was frequently associated with autoimmune thyroid disease is not clear, but T cell maturation defects and overexpression of chromosome 21 products in Down's syndrome have been suggested. A 19-year-old female was transferred because of generalized weakness. She was born to a 42-year-old mother, She had been suffered from heat intolerance, weight loss, palpitation, dyspnea on exertion and neck swelling and had intermittently taken some medication since her age 9. She had mental retardation (IQ 41) and underdeveloprnent. Exophthalmos, upward-outward slant of palpabral fissures, epicanthal folds, lowset ears, and large, protruding, fissured tongue were identified. Short fifth middle phalanges, clinodactyly and small-sized interventricular septal defect were also detected. Thyroid gland was diffusely enlarged four times the normal size, firm in consistency and had a bruit. Serum T concentration was 7.8ug/dL, T2 306ng/dL, and TSH 0.01ulU/mL. She was positive for thyroid autoantibodies (antimicosomal antibody 1,867 IU/mL, antithyroglobulin antibody 106 IU/mL, and TBII 79.6%). Twenty-four hours radioactive iodine uptake was 64%. Chromosomal analysis with T cell culture stimulated by phytohemagglutinin revealed 47XX, 21 trisomy. Pituitary hormones except TSH were fully stimulated by combined pituitary stimulation. She was finally diagnosed as Down's syndrome with Graves' disease and controlled with use of methimazole.

Original Article

Serum Lipoprotein (a) and Lipid Concentrations in Patients with Subelinical Hypothyroidism.: Kyoung Ah Kim, Jae Hoon Chung, Yeun Sun Kim, Kyu Jeung Ahn, Eun Mi Koh, Young Ki Min, Myung Shik Lee, Moon Kyu Lee, Jong Hun Lee, Kwang Won Kim; J Korean Endocr Soc. 1997;12(1):11-17. Published online January 1, 2001

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Abstract PDF: BACKGROUND
Overt hypothyroidism is well-known cause of secondary hyperlipidemia and atherosclerosis. However, there have been dissenting reports of abnormalities in serum lipid concentrations in patients with subclinical hypothyroidism (SH). Recently, it has been reported that serum Lp (a) concentration, an independent risk factor of atherosclerosis, was increased in patients with SH. Therefore, we analyzed serum Lp (a) and other lipid concentrations to investigate whether they are increased in patients with SH and the correlation between serum Lp (a) and TSH concentrations. METHODS: We undertook this study in 53 patients with SH (TSH > 6 uiU/ml) and 197 age-and sex-matched healthy control subjects, They had no abnormalities in liver function, BUN, creatinine, fasting blood glucose, urinalysis, and past medical histories. Serum T3, T4, and TSH concentrations were measured by RIA using commercial kits. Serum concentrations of Lp (a), total cholesterol, triglyceride (TG), and HDL cholesterol (HDL-C) were measured by rate nephelometry and enzyme assay, respectively. RESULTS: There were no significant differences of serum Lp (a), total cholesterol, LDL cholesterol, TG, and HDL-C concentrations in 53 patients with SH and 197 control subjects (25.6+-3.8mg/dL vs. 25.4+-1.5mg/dL ; 204.0+-4.2mg/dL vs. 204.0+-2.4mg/dL ; 127.0+-3.9mg/dL vs. 125.0+-2.3 mg/dL ; 133.0+-8.5mg/dL vs. 130.0+-6.0mg/dL ; 50.0+-1.5mg/dL vs. 53.0+-0.9mg/dL). There was no correlation between Lp (a) and TSH concentrations in SH (r=0.12, p>0.05). CONCLUSION: Serum Lp (a) concentration as well as total cholesterol, LDL cholesterol, and TG was not increased in patients with SH. There was no correlation between serum Lp (a) and TSH levels in subclinical hypothyroidism.

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