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 Adrenal adenoma Associated with Pregnancy.: Jung Gyn Kim, Jang Sik Choo, Yang Kyu Lee, Bung Chul Han, Seung Bum Jin, Sang Gi Yang, Chang Sup Song, Me Gyung Sin; J Korean Endocr Soc. 1994;9(1):39-45. Published online November 6, 2019

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Abstract PDF: We experienced a case of aldosterone and cortisol secreting adrenal adenoma associated with pregnancy in a 23 year old female patient.The patient complained of severe thoraco-lumbar pain, weight gain, sweating, anxiety, and mild abdominal discomfort. On physical findings, hypertension, tachycardia, facial plethora, moon face, buffalo hump and truncal obesity were found.

Original Article

A Clinical Observation on Twelve Cases of Primary Aldosteronism.: Seung Chul Cho, Yong Soo Park, Hwon Gyum Park, Sung Hee Lee, Soon Gil Kim, Woong Hwan Choi, Yu Hern Ahn, Pa Jong Jung, Tae Wha Kim; J Korean Endocr Soc. 2004;19(2):194-202. Published online April 1, 2004

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Abstract PDF: BACKGROUND
Primary aldosteronism describes a group of disorders characterized by long-standing aldosterone excess, with suppressed renin activity, resulting in hypertension and hypokalemia. The protean clinical and biochemical characteristics of this syndrome have important implications regarding its pathophysiology and responsiveness to treatment. METHODS: The cases of 12 primary aldosteronisms, diagnosed at Hanyang University Hospital between 1996 and 1999, were reviewed. RESULTS: The 12 cases were composed of 9 aldosterone-producing adenoma, 2 adrenal hyperplasia and a case of idiopathic hyperaldosteronism. There were 9 women and 3 men. The mean age was 46 yrs (range, 23 to 64 yrs). At the initial visit, the mean blood pressure was 160+/-26/104+/-14 mmHg, and one case of idiopathic hyperplasia had normal blood pressure. The mean serum K+ level was 2.6+/-0.5 mEq/L (range, 1.5 to 3.5 mEq/L). The mean plasma renin activity and plasma aldosterone concentration were 0.4+/-0.4 ng/ml/hr (range, 0.2 to 1.6 ng/ml/hr) and 407.5+/-199.8 pg/mL (range, 225 to 800 pg/mL), respectively. Different steps of diagnostic modalities were applied for the preoperative differential diagnosis. All patients, with the exception of the one with idiopathic hyperaldosteronism, were managed by a unilateral laparoscopic adrenalectomy, as they were all diagnosed under the impression of adrenal adenomas. Ultimately, 9 cases were proven to have adrenal adenomas. One hypertensive case, with hypokalemia, had adrenal hyperplasia, and the case with normotension was found to have adrenal nodular hyperplasia from the pathology. The size of the tumors ranged from 1.4 to 2.4 cm in diameter. Among the 11 cases that underwent an adrenalectomy, the blood pressures in 6 cases normalized after the operation, while the other 5, including the one with unilateral hyperplasia, were still in need of antihypertensives for the control of elevated blood pressures, even after the operation. The other case of idiopathic hyperaldosteronism was managed by the prescription of spironolactone. CONCLUSION: From these, it can be suggested that the clinical diversity of the syndrome, especially in the pathophysiology and response to operation, awaits the development of a better preoperative lateralization procedure

Case Reports

A Case of Cushing's Syndrome Complicating Pregnancy Adrenalectomized after Delivery.: Jeong Soo Cho, Jae Seok Jeon, Kyu Jeong Ahn, Byoung Joon Kim, Sung Ho Kwon, Eun Gyu Lee, Won Wook Choi, Sang Won Park, Won Mee Lee; J Korean Endocr Soc. 2003;18(5):489-495. Published online October 1, 2003

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Abstract PDF: Pregnancy is rare in women with Cushing's syndrome, as the associated infertility is related to excess cortisol and/or androgen. However, approximately 100 such cases have been reported, with 50% due to an adrenal cortical adenoma. Establishing a diagnosis and cause can be difficult. Clinically, striae, hypertension and gestational diabetes are common features in pregnancy, with hypertension and diabetes being the most common signs of Cushing's syndrome in pregnant women. Furthermore, biochemically, a normal pregnancy is associated with a several fold increase in plasma cortisol, as the increased cortisol production rate also increases the cortisol binding protein. Untreated, the condition results in high maternal and fetal morbidity and mortality. An adrenal or pituitary adenoma should be excised, but a metyrapone, which is not teratogenic, has been effective in controlling many cases of excess cortisol. Here, a case of Cushing's syndrome, complicating a pregnancy due to an adrenal cortical adenoma, with thorough obstetric and medical management, including a metyrapone, which was adrenalectomized after delivery, is reported.

A Case of Adrenocortical Adenoma Causing Cushing's Syndrome with Contralateral Nonfunctioning Adenoma.: Sun Young Kyung, Hye Sook Hahn, Hyo Joong Yoon, Young Ha Hwang, Chan Jong Seo, Yeon Sil Jeong, Hong Kyu Kim, Hye Young Park, Hyung Sik Kim, Jeong Nam Lee, Seung Yeon Ha, Moon Ho Kang; J Korean Endocr Soc. 2002;17(2):286-291. Published online April 1, 2002

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Abstract PDF: We report the case of a 43-year-old woman with Cushing's syndrome showing bilateral adrenococortical adenomas. We performed bilateral selective adrenal vein samplings. Hypersecretion of cortisol on the left sided adrenal tumor was observed, but no evidence of cortisol hypersecretion from the adrenal tumor on the right side was observed. The left adrenal tumor was resected selectively, but the right adrenal gland was reserved. The left adrenal tumor was histologically diagnosed as a adrenal adenoma without any evidence of nodular hyperplasia. Following the resection of the left adrenal gland, no cortisol hypersecretion from the remaining adrenal tumor on the right side was observed until now, suggesting that a selective adrenalectomy of functioning adenoma may be an acceptable treatment modality.

A Case of Adrenalectomy after Preterm Delivery in Cushing's Syndrome of Third Trimester Pregnant Woman.: Kwang Sik Song, Jae Kyung Hwang, Ki Tak Ju, Hang Jin Lee, Suk Ho Song, Ki Young Lee, Chan Jong Suh, Hong Kyu Kim, Hyu Young Park, Dal Mo Yang, Young Ha Oh, Moon Ho Kang; J Korean Endocr Soc. 2001;16(1):134-139. Published online February 1, 2001

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Abstract PDF: A renin- or angiotensin-II responsive aldosterone producing tumor is a rare cause of primary hyperaldosteronism. This tumor can be identified by tests that show that the aldosterone producing adrenal tumor is not fully autonomous. In other words partially it is responsible for the stimulation of aldosterone secretion that results aldosterone levels in an increase in serum in response to the upright posture and spironolactone treatment. Furthermore, the urinary 18-hydroxycortisol level is within the normal range. Because of different responses to surgical removal, the differential diagnosis of the causes of primary aldosteronism can't be overemphasized even for rare causes of primary aldosteronism such as unilateral nodular hyperplasia or a renin-responsible aldosterone producing tumor. We should consider renin or angiotensin-II responsive adrenal adenoma in the differential diagnosis of primary aldosteronism when biochemical data shows atypical results. Here we present the first case in Korea of a renin-responsive aldosterone producing adrenal adenoma which was fully accessible and was successfully treated by surgical removal. Also, sampling for aldosterone secretion just above the insertion site in the left renal vein before surgery showed a suspiciously abberant left adrenal vein drainage into the IVC, This was very helpful information during adrenal vein ligation in laparoscopic adrenalectomy.

Renin-responsive Adrenal Adenoma with a Suspicious Abberant Adrenal Vein.: Ji Hyeon Ju, Woo Lee Jung, Jung Min Lee, Kun Ho Yoon, Bong Yun Cha, Ho Young Son, Dong Gu Kim, Eung Kook Kim, Byung Ki Choi; J Korean Endocr Soc. 2001;16(1):123-129. Published online February 1, 2001

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Abstract PDF: A renin- or angiotensin-II responsive aldosterone producing tumor is a rare cause of primary hyperaldosteronism. This tumor can be identified by tests that show that the aldosterone producing adrenal tumor is not fully autonomous. In other words partially it is responsible for the stimulation of aldosterone secretion that results aldosterone levels in an increase in serum in response to the upright posture and spironolactone treatment. Furthermore, the urinary 18-hydroxycortisol level is within the normal range. Because of different responses to surgical removal, the differential diagnosis of the causes of primary aldosteronism can't be overemphasized even for rare causes of primary aldosteronism such as unilateral nodular hyperplasia or a renin-responsible aldosterone producing tumor. We should consider renin or angiotensin-II responsive adrenal adenoma in the differential diagnosis of primary aldosteronism when biochemical data shows atypical results. Here we present the first case in Korea of a renin-responsive aldosterone producing adrenal adenoma which was fully accessible and was successfully treated by surgical removal. Also, sampling for aldosterone secretion just above the insertion site in the left renal vein before surgery showed a suspiciously abberant left adrenal vein drainage into the IVC, This was very helpful information during adrenal vein ligation in laparoscopic adrenalectomy.

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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Abstract 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.

A Case of Black Adrenocortical Adenoma Causing Cushing's Syndrome with Contralateral Nonfuncioning Adenoma.: Do Joon Park, Kyung Soo Park, Kyung Jae Nam, Sung Yeon Kim, Bo Yeon Cho, Hong Gyu Lee, Yeo Kyu Yoon, Seung Keun Oh; J Korean Endocr Soc. 1999;14(2):410-417. Published online January 1, 2001

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Abstract PDF: We report herein the case of a 38-year-old woman with Cushings syndrome caused by bilateral adrenocortical adenomas. The adrenal tumor on the left side hypersecreted cortisol and no findings of cortisol hypersecretion from the adrenal tumor on the right side were observed on bilateral adrenal vein samplings. Both adrenal tumors were resected and histologically without any findings of nodular hyperplasia. The left adrenal tumor was histologically diagnosed as a so-called black adenoma. These data imply that the adrenal adenomas developed primarily from the adrenal gland itself, and that one of the tumors was well differentiated and secreted excess hormones, while the other remained in cell proliferation without hypersecretion.

Unolateral Aldosterone-producing Adenoma with a Contralateral Black Adenoma.: Eun Young Oh, Myung Shik Lee, Young Hee Lim, Soo Jung Kang, Jung Hak Chun, Byoung Joon Kim, Jae Hoon Chung, Yong Ki Min, Moon Kyu Lee, Kwang Won Kim, Jung Hyun Yang; J Korean Endocr Soc. 1999;14(1):177-182. Published online January 1, 2001

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Abstract PDF: Primary aldosteronism due to unilateral adenoma is a rare cause of surgically curable hypertension. Bilateral adrenal mass has occasionally been reported in this syndrome, and bilateral aldosterone-producing adenoma (APA), or bilateral adrenal nodular hyperplasia have been demonstrated in some cases. However, another possibility is the coexistence of a unilateral APA with a contralateral benign or metastatic nonfunctioning mass, because adrenal tumors are frequently found at autopsy or incidentally detected during abdominal morphological evaluation in patients without adrenal dysfunction. A 39 year-old woman presented with hyperaldosteronism, suppressed renin levels, and bilateral adrenal mass on adrenal CT scanning. Selective adrenal venous sampling was unsuccessful in demonstrating concentration gradient of aldosterone. Postoperative measurement of hormone content in the tumor extract revealed unilateral aldosteron-producing adenoma with contralateral nonfunctioning black adenoma, Determination of hormone content in the tumor extract could be useful for the discrimination of functioning and nonfunctioning endocrine tumors, particularly in case of multiple tumors.

A Case of Cushing's Syndrome in Pregnancy due to Adrenal Adenoma.: Hyung Joon Yoo, Sung Hee Ihm, Sung Woo Park, Hae Sung Yim, Yong Tae Kim, Chul Hee Park, Hyun Kyu Kim, Doo Man Kim, Jae Myoung Yoo, Moon Ki Choi; J Korean Endocr Soc. 1998;13(2):264-270. Published online January 1, 2001

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Abstract PDF: Cushings syndrome in pregnancy is rare. This is explained by the syndromes association with amencerhea, infertility and abortions. Matemal and fetal risks increase markedly when pregnancy does occur in woman with hypercortisolism. Since pregnant women without Cushings syndrome develop some features of Cushings syndrome, such as hypertension, hyperglycemia and striae, a high index of clinical suspician must be maintained to prevent delay in diagnosis. The physiologic changes in adrenocorticostemid metabolism during pregnancy further complieate the diagnosis. We describe a case of Cushings syndmme in pregnancy secondary to an adrenal cortical adenoma which was diagnosed immediately after a preterm delivery in 24-year-old woman with preeclampsia.

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