TY - JOUR
T1 - Coexistence of three distinct adrenal tumors in the same adrenal gland in a patient with primary aldosteronism and preclinical Cushing's syndrome
AU - Okura, Takafumi
AU - Miyoshi, Ken Ichi
AU - Watanabe, Sanae
AU - Kurata, Mie
AU - Irita, Jun
AU - Manabe, Seiko
AU - Fukuoka, Tomikazu
AU - Higaki, Jitsuo
AU - Sasano, Hironobu
PY - 2006/6/1
Y1 - 2006/6/1
N2 - A 62-year-old woman was admitted to our hospital because of hypokalemia. Physical examination revealed no signs of excessive adrenocortical steroid production, as are found in Cushing's syndrome. Her plasma renin activity (PRA) was suppressed (0.10ng/ml per h), and her serum aldosterone level was high (30.0ng/dl). PRA was not increased after a renin-releasing test. Her plasma adrenocorticotropic hormone (ACTH) level was low (<5pg/ml), but her serum cortisol level was normal (21.0μg/dl). Administration of 8mg dexamethasone did not suppress her plasma cortisol level. Finally, she was diagnosed with clinical primary aldosteronism associated with preclinical Cushing's syndrome. Magnetic resonance image revealed three sequential nodular masses (each 15mm × 15mm) in the right adrenal gland. A right adrenalectomy was performed by endoscopy. The three removed tumors appeared to have different characteristics. Microscopic examination revealed that the upper and lower tumors were adrenocortical adenomas, and the middle tumor was a black adenoma. Immunohistochemical staining for the enzymes involved in cortisol biosynthesis suggested that the upper tumor secreted aldosterone, whereas either or both of the two other tumors secreted cortisol. Surprisingly, at 33 years of age, she had been diagnosed with Cushing's syndrome, due to a cortisol-producing adrenocortical adenoma, and she had received a left adrenalectomy. Clinically and pathophysiologically, this was a very rare case.
AB - A 62-year-old woman was admitted to our hospital because of hypokalemia. Physical examination revealed no signs of excessive adrenocortical steroid production, as are found in Cushing's syndrome. Her plasma renin activity (PRA) was suppressed (0.10ng/ml per h), and her serum aldosterone level was high (30.0ng/dl). PRA was not increased after a renin-releasing test. Her plasma adrenocorticotropic hormone (ACTH) level was low (<5pg/ml), but her serum cortisol level was normal (21.0μg/dl). Administration of 8mg dexamethasone did not suppress her plasma cortisol level. Finally, she was diagnosed with clinical primary aldosteronism associated with preclinical Cushing's syndrome. Magnetic resonance image revealed three sequential nodular masses (each 15mm × 15mm) in the right adrenal gland. A right adrenalectomy was performed by endoscopy. The three removed tumors appeared to have different characteristics. Microscopic examination revealed that the upper and lower tumors were adrenocortical adenomas, and the middle tumor was a black adenoma. Immunohistochemical staining for the enzymes involved in cortisol biosynthesis suggested that the upper tumor secreted aldosterone, whereas either or both of the two other tumors secreted cortisol. Surprisingly, at 33 years of age, she had been diagnosed with Cushing's syndrome, due to a cortisol-producing adrenocortical adenoma, and she had received a left adrenalectomy. Clinically and pathophysiologically, this was a very rare case.
KW - Adrenocortical adenoma
KW - Black adenoma
KW - Lipofuscin
KW - Preclinical Cushing's syndrome
KW - Primary aldosteronism
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U2 - 10.1007/s10157-006-0413-z
DO - 10.1007/s10157-006-0413-z
M3 - Article
C2 - 16791399
AN - SCOPUS:33745499258
VL - 10
SP - 127
EP - 130
JO - Clinical and Experimental Nephrology
JF - Clinical and Experimental Nephrology
SN - 1342-1751
IS - 2
ER -