Endocrinology and Metabolism

Case Reports

A Case of Giant Adrenal Adenoma Presenting Primary Aldosteronism.: Ji Hyun Lee, Bong Soo Cha, Moon Suk Nam, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Hyung Chan Suh, Young Hwa Choi, Jae Min Park, Jung Soo Park, Soon Won Hong, Dong Hwan Shin; J Korean Endocr Soc. 1996;11(3):348-354. Published online November 7, 2019

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Abstract PDF: Primary aldosteronism is a syndrome chracterized by hypokalemic alkalosis and hypertension. Small sized adrenal cortical adenomas have been the major cause of this syndrome in most of the patients. However, if the adrenal mass is larger than 6cm in diameter and with irregular consistency, malignancy is more favored. We experienced a patient who had a giant adrenal adenoma with primary aldosteronism. A 24-year-old female presented with hypertension, hypokalemia, low plasma renin, and high plasrna aldosterone levels, was found to have a 6×5.5×5 cm sized left adrenal tumor by MRI. Her clinical laboratory feature did not revealed any evidence of Cushing's syndrome or pheochromocytoma. Preoperatively adrenal carcinoma presenting pure adrenal aldosteronism was suspected due to large size and heterogenous signal character of the adrenal mass in radiologic study. At operation well encapsulated, round giant adrenal tumor weighing 65gm(4.5×4×4 cm) was removed. There was no evidence of metastasis with return of adrenal function to normal after surgery. Benign adrenal adenoma was confirmed by the gross morphology and the histologic features.

A Case of Adult Fanconi Syndrome with Hypophosphatemic Osteomalacia.: Ji Hyun Lee, Young Sup Byun, Bong Soo Cha, Moon Suk Nam, Young Duk Song, Sung Kil Lim, Kyung Rae Kim, Hyun Chul Lee, Kap Bum Huh, Jin Kim, Jong In Yook; J Korean Endocr Soc. 1996;11(1):93-101. Published online November 7, 2019

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Abstract PDF: The Fanconi syndrome is characterized by generalized disturbance of tubular function. It leads to excessive losses of amino acids, glucose, phosphate, bicarbonate, and other organic and inorganic substrates handled by the proximal tubules. The metabolic consequences are acidosis, hypophosphatemia, hypocalemia, dehydration, rickets, osteomalacia, osteoporosis, and growth retardation. This syndrome may either be congenital or acquired, primary or secondary. Acquired Fanconi syndrome may result from multiple myeloma, Wilsons disease, primary amyloidosis, light chain nephropathy, and heavy metal poisoning such as lead, mercury, and cadmium. A 33-year-old female presented with multiple bone pain, and progressive proximal muscle weakness for 15 months. The blood urea nitrogen, creatinine, calcium, phosphate, and uric acid were 12.1 mg/dL, 1.5 mg/dL, 8.4 mg/dL, 1.8 mg/dL, and 1.7 mg/dL, respectively. The urine volume, protein, calcium, phosphate, and creatinine clearance were 2,330 ml, 343.7 mg, 146 mg, 424 mg, and 44.6 ml/min, respectively in 24 hour collection urine study. The tubular reabsorption rate of phosphate was decreased. In arterial blood gas analysis study, pH was 7.348, bicarbonate was 17.6 mmol/L, which means metabolic acidosis. In chest X-ray, fracture was seen in eighth and ninth left ribs. The whole body bone scan revealed hot uptake at both first and second ribs, right third rib, both eighth and ninth ribs, left sacroiliac joint and right hip joint. Bone densitometry showed moderate osteopenia in spine and femur neck. After NE4Cl loading, the urine pH was decreased below 5.0 at two and third hour, which means proximal renal tubular acidosis. Amino acid such as, hydroxyproline, threonine, serine, asparagine, glutamine excreted much more than normal in 24 hour urine. Bone biopsy showed the presence of increased osteoid volume and osteoid seam width and marked decreased mineral appositional rate as evidence for osteomalacia. The patients symptoms, including bone pain and proximal muscle weakness, were relieved after supplement of calcitonin, Vitamin D and calcium carbonate. We report a case of Fanconi syndrome with hypophosphatemic osteomalacia with brief review of literature.

A Case of Anterior Cervical Lipoma Mimicking Diffuse Goiter.: Eun Jig Lee, Moon Suk Nam, Su Youn Nam, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Kyung Rae Kim, Jun Sik Na, Yee Hyun Nam, Jeon Hong Kang, Jung Ki Seo; J Korean Endocr Soc. 1995;10(4):418-423. Published online November 7, 2019

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Abstract PDF: Lipoma is a benign fatty tumor that can arise in any location of the body where fat is present. It is found most commonly in the upper half of the body, particularly the head and neck, shoulders, and back. A mass in the antero-inferior part of the neck may be initially thought to be thyroid masses and then other cervical masses should be considered. Ultrasongraphic examination of benign lipoma demonstrates solid and echogenic mass and may differentiate nonthyroid from thyroid masses. Although the location of tumors, its consistency, and its motion with deglutition, seperation from the thyroid on sonographic examination, all pointed to nonthyroidal origin, did not rule out a possible mass that isolated from the lobes of the thyroid. Fine needle aspiration and biopsy can provide clear answer.We herein report a case of anterior cervical mass in a 48-yr-old male patient presenting a non-tender, slightly hard and nodular mass slowly growing for several years and moved with swallowing, and diagnosed his case as benign lipoma using thyroid scan and ultrasonography. When we encounter patients with anterior neck mass, we should consider benign lipoma mimicking diffuse goiter.

Original Articles

Clinical and Endocrinologic Differences between Prolactinoma and Pseudoprolactinoma Proven by Immunohistochemical Study.: Jae Wha Jo, Eun Jig Lee, Moon Suk Nam, Su Youn Nam, Young Duk Song, Hyun Chul Lee, Kap Bum Huh, Tae Seung Kim, Sun Ho Kim, Kyung Rae Kim, Bong Soo Cha, Ji Hyun Lee, Sung Kil Lim; J Korean Endocr Soc. 1995;10(4):362-369. Published online November 7, 2019

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Abstract PDF: Hyperprolactinemia is the most common hypothalamo-pituitary disorder encountered in clinical endocrinology. Excluding the drug-induced hyperprolactinemia, the most common cause of this disorder is a pituitary tumor. Prolactinoma is mainly made up of prolactin-secreting cells but pseudoprolactinoma is tumor that does not secrete prolactin itself. The pseudoprolactinoma interrupts the flow of prolactin inhibiting factor, dopamine, from the hypothalamus through the pituitary stalk to the normal pituitary. The differentiation prolactinoma from pseudoprolactinoma is vitally important since true prolactinomas are most commonly responded well in terms of tumor shrinkage to medical treatment using dopamine agonist therapy, whereas pseudoprolactinomas do not. Thus surgical treatment is clearly indicated as first-line treatment if we know that a lesion is a pseudoprolactinoma. We compared prolactinoma with pseudoprolactinoma in clinical and endocrinologic characteristics of 48 cases after immunohistochemical diagnosis. We could not find any differential point of both tumors in clinical and radiological characteristics although some differences were exist. But we had found the relationship between the mean level of pretreatment serum prolactin and the presence of positive immunohistochemical stain for prolactin. The pretreatment serum prolactin level was significantly higher in patients with tumors showing many prolactin immunohistochemical staining cells than in those with none(p<0.05). When the pretreatment serum prolactin exceeded 100ng/ml, the tumors contain 94% of prolactin positive cells in stain. So, if the pretreatment serum prolactin exceeds 100ng/ml, we primarily suspect prolactinoma and medical treatment should be considered. If the pretreatment level below 100ng/ml, we suspect pseudoprolactinoma and surgical treatment should be considered.

Immunohistochemical Study of c - Myc, c - Fos and c - Jun Oncoprotein Expression in the Human Pheochromocytoma.: Jae Wha Jo, Eun Jig Lee, Moon Suk Nam, Kyung Rae Kim, Su Youn Nam, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Yong Hye Lee, Tae Seung Kim, Kwan Woo Lee; J Korean Endocr Soc. 1995;10(1):26-34. Published online November 6, 2019

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Abstract PDF: A large number of studies for genes involved in oncogenesis have been done during last decade. Over 20 oncogenes have been isolated characterized, and the oncogene expressions in human tumors have been examined. The proto-oncogenes of c-Myc, c-Fos and c-Jun, which modulate the transcription factors, have overexpressed in a variety of human cancers. Immunohistochemical method was used in this study to examine c-Myc, c-Fos and c-Jun oncoprotein expression in 31 patients with human pheochromocytoma 28(90.0%) were benign and 3(10.0%) malignant. C-Myc oncoprotein immunoreactivity was found in 24 cases(77.4%), c-Fos in 29(93.5%), and c-Jun in 25(80.6%). Twenty-one(67.7%) showed positive immunoreactivity for all these oncoproteins, six(19.4%) for 2 oncoproteins, 3 for one oncoprotein. Only 1 case showed negative immunoreactivity for all 3 oncoproteins. The oncoprotein immunoreactivity did not correlate with the amount of 24 hour urinary catecholamine excretion. Although the number of malignant pheochromocytomsa was not so many, most of them showed that the immunoreactivity for oncoprotein was more than 30 percent of tumor cells.The expression of c-Myc, c-Fos and c-Jun oncoprotein were frequently found in human pheochromocytoma. These results suggest that the oncoprotein expression may play an important role in tumorogenesis and proliferation of human pheochromocytoma.

Case Report

A Case of Idiopathic Juvenile Osteoporosis.: Moon Suk Nam, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Young Joon Weon, Bong Soo Cha, Ji Hyun Lee, Jung Ho Lee; J Korean Endocr Soc. 1994;10(3):278-283. Published online November 6, 2019

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Abstract PDF: Idiopathic juvenile osteoporosis is a rare disease of heterogenous etiology and occurs on children between the age of 8 and 15. Manifestations include bone pain, fractures in minimal trauma, reduced bone density at areas of new bone growth, and loss of height. It is important to exclude other causes of osteoporosis.We experienced a case of a 14 year old boy with idiopathic juvenile osteoporosis. He had suffered from pain in the back and difficulty on walking for two months. Radiologic finding of the thoracolumbar area of the spine showed generalized severe osteoporosis and multiple vertebral collapse. We could not find the causes of osteoporosis in biochemical study, bone marrow study, skin biopsy and hormonal study. He was treated with alphacalcidol and CaCO_3. After 4 month of initial management, his subjective symtoms were improved and we did not find any signs of progression of disease. On bone mineral density measured after 26 month, we observed markedly increased bone mineral density.We report our experience of follow up of this case and review with the disease reported in the literature.

Original Articles

Complication and Prognosis of Craniopharyngioma According to the Age of Onset.: Eun Jig Lee, Moon Suk Nam, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Kyung Rae Kim, Kun Hoon Song, Bong Soo Cha, Ji Hyun Lee; J Korean Endocr Soc. 1994;10(3):262-272. Published online November 6, 2019

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Abstract PDF: Craniopharyngioma is the most common tumor involving the hypothalamo-pituitary area in childhood and adolescence. Recently, we carried out collective review of 70 patients with craniopharyngioma treated from January 1980 to December 1994 in order to inverstigate the endocrine outcome and survival according to the age of onset.The following results were obtained:1) The male to female ratio was 1:1. Age at diagnosis ranged from 2 to 64 years(mean age: 23) with the greatest frequency in the 2nd decade of life(28.6%). Of the 70 cases, the first group, 27 cases were under the age of 15, and the other group, 43 cases were over 15 year-old.2) The most common symptom at diagnosis in both groups was headache. In the adult group, symptoms related to hypogonadism(amenorrhea, decreased libido, galactorrhea etc.) were not uncommon. The lag of time between onset of symptom and hospital visit ranged from 3 days to 156 months(mean: 20 months).3) The main site of tumor was suprasellar region in both groups. The most common CT finding in both groups was calcification in sella turcica.4) In pre-operative combined pituitary function test, the most common, abnormal responses were shown in growth hormone and thyroid stimulating hormone in both groups. In addition, prolactin frequently showed abnormal response in the adult group.In post-operative combined pituitary function test, more hormones tended to reveal abnormal response in the group treated with surgery plus radiation therapy.5) The operation by subtotal removal followed by radiation therapy was the most commonly used method in treatment of both groups. After treatment, panhypopituitarism was occurred more frequently in the group treated with RT after surgery than those treated with surgery alone, but the difference was not statistically significant(p=0.136 in childhood, 0.436 in adults). Except the cases with panhypopituitarism, the most commonly encountered endocrine abnormalities were growth retardation in the children group, and hypogonadism in adult. The recurrence was clinically observed in 11 cases. The recurrence rate were 11.1% in children, and 18.6% in adult respectively. The mean time from the initial treatment to recurrence was 23 months. There was no significant difference in recurrence rate between the group treated with RT after subtotal removal and the group treated with total removal(p=0.475).The overall five-year survival rate after treatment was 82.8%. According to the treatment modalities, the patients undergone RT after subtotal removal survived much longer than those treated with other modalities such as subtotal removal only or total removal, but the differences in survival were not statistically significant(Log rank test, p=0.0539).

The Effect of Tumor Necrosis Factor - α on Extra Thyroidal Conversion of T4 to T3 in Slices and Extracts of Porcine Liver and Kidney.: Jae Wha Jo, Moon Suk Nam, Su Youn Nam, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Kyung Rae Kim, Eun Jig Lee; J Korean Endocr Soc. 1994;10(3):242-248. Published online November 6, 2019

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Abstract PDF: Abnormal thyroid function is seen in a wide variety of patients with acute and chronic nonthyroidal-illness, consisting of reduction in both thyroid function and peripheral T_4 to T_3 conversion including decreased serum thyroxin(T_4), decreased triiodothyronine and normal or slightly increased serum TSH. Recently increasing awareness of the role tumor necrosis factor-alpha(TNF-alpha) in systemic illness led us to consider a possible contribution of TNF-alpha to thyroid dysfunction in this setting. So we hypothesized that TNF-alpha might affect peripheral conversion of T_4 to T_3. We, therefore, explored the effects of TNF-alpha in slices and extracts of porcine liver and kidney. Thyroxine(T_4, 5ug/ml) was incubated in 0.15M phosphate buffer(PH 7.4) with slices(approximately 0.3 g-equivalent of tissue) and extracts(approximately 0.23 g-equivalent of tissue) of porcine liver and kidney with various concentrations(0-500pg/ml) of recombinant human TNF-alpha for 2 hours at 37 degree, and the T_3 generated during incubation was measured by radioimmunoassay of an ethanol extracts of the extracts and the homogenates of the slices. The slices produced more T_3 than extracts and the kidney was more active than the liver. TNF-alpha inhibited T_4 to T_3 conversion in dose-dependent manner in slices and extracts of porcine liver and kidney. Our data suggest that TNF-alpha have inhibitory effect of T_4 to T_3 conversion in liver and kidney(J Kor Soc Endocrinol 10: 242-248, 1995).

The Effect of Tumor Necrosis Factor - α on the Thyroglobulin Synthesis and TSH Action in Cultured Porcine Thyroid Cells.: Jae Wha Jo, Eun Jig Lee, Moon Suk Nam, Su Youn Nam, Young Duk Song, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Kyung Rae Kim; J Korean Endocr Soc. 1994;10(3):220-228. Published online November 6, 2019

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Abstract PDF: Abnormal thyroid function is seen in a wide variety of patients with acute and chronic nonthyroidal-illness, consisting of reduction in both thyroid function and peripheral T_4 to T_3 conversion including decreased serum thyroxin(T_4), decreased triiodothyronine and normal or slightly increased serum TSH. Recently increasing awareness of the role of tumor necrosis factor-alpha(TNF-alpha) in systemic illness led us to consider a possible contribution of TNF-alpha to thyroid dysfunction in this setting. So we hypothesized that TNF-alpha might affect the function of the thyroid gland. We, therefore, explored the effects of TNF-alpha on the cultured porcine thyroid cells in TSH-stimulated and TSH-nonstimulated conditions. Primarily cultured porcine thyroid cells were incubated with various concentrations(-500pg/ml) of recombinant buman TNF-alpha and bTSH(1mu/ml), with measurement of secreted thyroglobulin(Tg) and cyclic AMP(c-AMP) as the end points of stimulation, and produced intracellular Tg by pulse-labelling. TNF-alpha significantly inhibited TSH-stimulated intracellular Tg synthesis and extracellar Tg secretion at 200 and 500pg/ml concentration. TNF-alpha didn't affect c-AMP production at any concentration tested. So we conclude that increased in serum TNF-alpha may be responsible for reduced thyroid function in patients with acute and chronic nonthyroidal-illness.

Endocrinological and Morphological Characteristics of Clinically Nonfunctioning Pituitary Adenoma.: Jae Wha Jo, Moon Suk Nam, Hyun Chul Lee, Tae Seung Kim, Kyu Chang Lee, Hyun Joo Jung; J Korean Endocr Soc. 1994;9(3):200-212. Published online November 6, 2019

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Abstract PDF: Forth-nine patients with clinically nonfunctioning pituitary adenomas were evaluated clinically, endocrinologically and morphologically in this study.The results obtained were as follows.1) The mean age was 47.1 years(range 23 to 76 years), and 22 were male(44.9%) and 27(55.1%) female.2) The major clinical manifestations of male patients were visual disturbance(72.7%), headache(54.5%), loss of libido(45.5%), but those of female visual disturbance(59.6%), headache(48.1%), amenorrhea(48.1%), loss of body hair(25.9%), and galactorrhea(22.2%).3) All were macroadenomas evaluated by CT scan, and in the male patients 16(72.7%) were grade III and 6(27.3%) grade IV by Hardy classification, and in the female patients 6(22.2%) were grade II, 12(44.4%) grade III, and 9(33.3%) grade IV.4) The elevation of serum prolactin were observed 7(31.8%) out of male, and 24(88.9%) of female.5) Combined stimulation test revealed that GH insufficiency was 89.6%, ACTH 58.9%, LH 58.7%, FSH 51.1 %, and TSH 50.0% and hormone insufficiency more than 4 pituitary hormone was 54.2%.6) Prolactin response to TRH decreased in 12(70.6%) of 17 patients with normal basal prolactin, and 19(76.0%) of 25 with elevated prolactin.7) Immunohistochemistry revealed that null cell adenoma was 57.1%, gonadotrope adenoma 26.5%, plurihormonal adenoma 8.0%, silent corticotrope adenoma 4.0%, thyrotrope adenoma(2.0%), and lactotrope adenoma(2.0%).8) The ultrastructural characteristics examined by electron-microscopy were similar despite of immunohistochemical differences.In summary, the prevalance of clinically nonfunctioning pituitary adenoma was middle aged men and women, and their main symptoms were visual disturbance and headache. Hyperprolactinemia and pituitary hormone insufficiency more than 4 hormone were observed commonly. Most of them were null cell adenoma and gonadotrope adenoma examined by immunohistochemistry. Further study using modern techniques: cell culture, subunit-immunostaining. And Northern blot analysis of mRNA for pituitary hormone or subunit, will be needed to clarify null cell adenomas.

In Situ Hybridization Analysis of Human Growth Hormone and Prolactin Secreting Pitultary Adenomas.: Jae Wha Jo, Eun Jig Lee, Moon Suk Nam, Kyung Rae Kim, Sung Kil Lim, Hyun Chul Lee, Kap Bum Huh, Tae Seung Kim, Sun Ho Kim, Joong Uhn Choi, Kyu Chang Lee, Hyun Joo Jung, Sang Seop Chung; J Korean Endocr Soc. 1994;9(2):82-92. Published online November 6, 2019

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Abstract PDF: A non-isotopic in situ hybridization method with biotin-labelled oligonucleotide probes was used to examine growth hormone(GH) and prolactin(PRL) gene expression in 32 patients with pituitary adenomas; 13 were prolactinomas, 8 GH secreting adenomas, and 11 mixed GH and PRL secreting adenomas.Positive immunostaining for GH was found in all patients with GH secreting adenomas, and mixed GH and PRL secreting adenomas. Positive immunostaining for PRL was found in all patients with prolactinomas and 9(81.8%) of 11 mixed GH and PRL secreting adenomas, 5(62.5%) of 8 GH secreting adenomas. Immunohistochemistry revealed that 13 were lactotrope adenomas, 5 somatotrope adenomas, and 14 GH and PRL cell adenomas.In situ hybridization revealed that GH mRNA expression was found in all the patients with somatotrope adenomas and GH and PRL cell adenomas, and 6(46.1%) of 13 lactotrope adenomas. PRL mRNA expression was 100% in lactotrope and GH and PRL cell adenomas, and 4(80.0%) of 5 somatotrope adenomas.The patients with a clinical diagnosis of acromegaly had detectable PRL mRNA in their neoplasm and it is suggested that the PRL cells in the adenomas did not result from dedifferentiation, but from the neoplastic stimulus for some mixed tumors probably occurred in cells previously committed to produce PRL and GH. In lactotrope adenomas, the PRL cells of the patients without expression of GH mRNA may be arised from cells programmed to secrete PRL or precussor PRL cells rather than from mixed GH-PRL cells. The finding that some patients produced mRNA detectable by in situ hybridization, but no hormone detectable by immunohistochemistry within tumor was suggested of a silent adenoma.These observations indicated that in situ hybridization studies may improve the classification of pituitary adenomas and may provide a precise knowledge of the biology of these neoplasms.

Obesity and Metabolism
Metformin-Associated Lactic Acidosis: Predisposing Factors and Outcome: Min Ju Kim, Ju Young Han, Jun Young Shin, Shin Il Kim, Jeong Min Lee, Seongbin Hong, So Hun Kim, Moon Suk Nam, Yong Seong Kim; Endocrinol Metab. 2015;30(1):78-83. Published online March 27, 2015; DOI: https://doi.org/10.3803/EnM.2015.30.1.78

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Abstract PDF PubReader

Background

Metformin is considered the first choice oral treatment for type 2 diabetes patients in the absence of contraindications. Rarely, life-threatening complications associated with metformin treatment are seen in some patients with underlying diseases. The aim of this study was to further investigate the clinical profiles and risk factors for metformin-associated lactic acidosis (MALA) and the treatment modalities according to survival.

Methods

To identify MALA, we performed a retrospective study in seven diabetic patients who were taking metformin and had been diagnosed with lactic acidosis at Inha University Hospital between 1995 and 2012. For each patient, we recorded the age, sex, daily metformin dosage, laboratory test results, admission diagnosis, and risk factors. Also, concurrent conditions, treatment modalities, and outcomes were evaluated.

Results

Six patients had risk factors for lactic acidosis before admission. All patients had renal impairment on admission as a precipitating risk factor. Five patients survived and two patients died despite early renal replacement therapy. Older patients tended to have a poorer prognosis.

Conclusion

Renal function must be monitored in elderly type 2 diabetes mellitus patients with underlying diseases and conditions causing renal impairment who begin metformin treatment. Accurate recognition of MALA and initiation of renal replacement are essential for treatment.

Citations

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Case Reports

A Case of Spontaneous Rupture of Adrenal Pheochromocytoma.: Eun Joo Kim, Mie Jin Lim, Byoung Wook Bang, Hyun Joo Park, Seong Bin Hong, Moon Suk Nam, Yong Seong Kim, Suk Jin Choi; J Korean Endocr Soc. 2006;21(5):424-427. Published online October 1, 2006; DOI: https://doi.org/10.3803/jkes.2006.21.5.424

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Abstract PDF

Although the spontaneous rupture of a pheochromocytoma is rare, it can be fatal if not promptly diagnosed. Hemorrhagic necrosis of the tumor will require an emergency operation, as this causes intraperitoneal hemorrhaging. Excessive vasoconstriction, tachycardia and labile blood pressure are the signs of a ruptured pheochromocytoma. The standard diagnostic tool of such a condition is an abdominal computed tomographic scan. Although the exact mechanism of the rupture is still unknown, a hemorrhage inside the tumor or a high intra-cortical pressure, due to rapid growth of the tumor, is known to play a major role in the pathogenesis. Herein is reported the case of a 53-year old male patient with a pheochromocytoma and an acute abdomen. The CT scan showed a left adrenal gland mass, with a hemorrhage in the left perirenal space. The patient underwent an emergency adrenalectomy, with the final histopathological diagnosis being that of a pheochromocytoma. Prompt recognition and early surgical intervention can improve outcome.

Citations

Citations to this article as recorded by

Ruptured functioning adrenal tumour, atypical presentation with renal colic and hypertension
Amr Elmoheen, Mohamed Yousry, Ahmed Elmesery, Khalid Bashir
BMJ Case Reports.2020; 13(12): e236050. CrossRef
Spontaneous ruptured pheochromocytoma: an unusual case report and literature review
Ye Seob Jee
Annals of Surgical Treatment and Research.2017; 93(3): 170. CrossRef

A Case of Bilateral Macronodular Adrenocortical Hyperplasia Accompanied by Hyperresponsiveness to Vasopressin.: Deok In Kim, Seung Hee Lee, Eun A Kim, Hwi La Park, Gyeong Ug Lee, Seong Bin Hong, Moon Suk Nam, Seok Hwan Shin, Yong Seong Kim; J Korean Endocr Soc. 2005;20(4):390-394. Published online August 1, 2005; DOI: https://doi.org/10.3803/jkes.2005.20.4.390

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Abstract PDF: Cushing's syndrome associated with nodular adrenal hyperplasia glands is divided into 4 main categories: adrenal adenoma, adrenal carcinoma, primary pigmented nodular adrenal dysplasia and macronodular adrenal hyperplasia(MAH). The mechanism of bilateral MAH, when ACTH is suppressed, was previously unknown, and referred to as being "autonomous". Recently, several reports have shown MAH to be under the control of ectopic or eutopic membrane hormone. Here, a case of Cushing's syndrome, caused by bilateral MAH, is reported. A 62-year-old woman presented with Cushingoid features, hypertension and diabetes mellitus. In her case, abnormal adrenal stimulation of cortisol secretion in response to exogenous vasopression stimulation was shown. Her urine free cortisol was 726.0microgram/dL, which was not suppressed after administration of high-dose dexamethasone. Her plasma cortisol level was elevated, but without circadian rhythm. ACTH was undetectable. An abdomen CT scan demonstrated bilaterally enlarged multinodular adrenal glands. A Sella MRI revealed no alteration of the pituitary gland. The patient underwent a laparoscopic bilateral adrenalectomy. Histological examination revealed bilateral macronodular hyperplasia. After having recovered, the patient showed progressive regression of the Cushingoid status.

A Case of Isolated Familial Somatotropinoma.: Hwi Ra Park, Eun A Kim, Mei Hua Jiang, Chang Soo Jang, Kyoung Wook Lee, Seong Bin Hong, Eun Young Kim, Myung Kwan Lim, Moon Suk Nam, Yong Seong Kim; J Korean Endocr Soc. 2004;19(4):398-405. Published online August 1, 2004

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Abstract PDF: The familial occurrence of a pituitary adenoma associated with multiple endocrine neoplasia (MEN) type 1 or Carney complex is a well-recognized entity. However, an isolated familial somatotropinoma is a rare inherited disease, which is characterized by clustering of a somatotrophic adenoma and acromegaly or gigantism in a family, but without other manifestations of MEN type 1, with only 68 cases, in 28 families, described in the literature. The mode of inheritance is autosomal dominant, with incomplete penetration, but the genetic background of these pituitary adenomas remains unknown. A family exists where both the father and son were affected. Endocrinological investigations confirmed hypersecretion of GH and IGF-1, and the pituitary adenomas were identified by magnetic resonance image in both cases. There was no symptom of MEN type 1 or other form of endocrine dysfunction. Herein is reported a case of an isolated familial somatotropinoma in Korea, with a review of the literature

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