Difficult-to-control hypertension due to bilateral aldosterone-producing adrenocortical microadenomas associated with a cortisol-producing adrenal macroadenoma.
The patient was a 54-year-old woman who developed a right adrenal tumour, Cushingoid features, elevated levels of cortisol that were not suppressed by 1 nor 8 mg of dexamethasone, and suppression of adrenocorticotropin (ACTH) during treatment for severe hypertension. Computed tomography (CT) revealed a right adrenal tumour and an atrophic left adrenal gland. In addition, elevated plasma aldosterone concentration (PAC) and suppressed plasma renin activity (PRA) with an aldosterone-to-renin ratio of 128 (ng per 100 ml per ng ml⁻¹ h⁻¹) suggested aldosterone excess. Urinary excretion of aldosterone was relatively high, and the captopril and rapid ACTH tests resulted in no response of PRA and exaggerated increase in PAC, respectively. ACTH-loaded adrenal venous sampling showed bilateral excess of aldosterone with right predominance of cortisol. Right laparoscopic partial adrenalectomy (ADX) and immunohistochemical analysis showed both a cortisol-producing adenoma and an aldosterone-producing microadenoma (microAPA) within the attached adrenal, which had not been detected by CT preoperatively. After the right partial ADX, her blood pressure, aldosterone level and suppressed PRA remained unchanged. Subsequently, laparoscopic total left ADX was performed. Two microAPAs with paradoxical hyperplasia were revealed within the apparently atrophic left adrenal gland. Soon after the second surgery, her blood pressure normalized without requiring any anti-hypertensive medication.
Morimoto
R
R
Division of Nephrology, Endocrinology, and Vascular Medicine, Department of Medicine, Tohoku University Graduate School of Medicine, Sendai, Miyagi, Japan.
Kudo
M
M
Murakami
O
O
Takase
K
K
Ishidoya
S
S
Nakamura
Y
Y
Ishibashi
T
T
Takahashi
S
S
Arai
Y
Y
Suzuki
T
T
Sasano
H
H
Ito
S
S
Satoh
F
F
D000302:Adrenal Cortex / Q000000981:diagnostic imaging / Q000473:pathology; D000306:Adrenal Cortex Neoplasms / Q000097:blood / Q000150:complications / Q000000981:diagnostic imaging / Q000601:surgery; D000315:Adrenalectomy; D018246:Adrenocortical Adenoma / Q000097:blood / Q000150:complications / Q000000981:diagnostic imaging / Q000601:surgery; D000450:Aldosterone / Q000097:blood*; D003480:Cushing Syndrome / Q000097:blood / Q000000981:diagnostic imaging / Q000209:etiology / Q000628:therapy; D005260:Female; D006801:Humans; D006854:Hydrocortisone / Q000097:blood*; D006929:Hyperaldosteronism / Q000097:blood / Q000000981:diagnostic imaging / Q000209:etiology / Q000628:therapy; D006973:Hypertension / Q000097:blood / Q000209:etiology* / Q000503:physiopathology / Q000628:therapy; D008875:Middle Aged; D014057:Tomography, X-Ray Computed; D016896:Treatment Outcome
D000450:Aldosterone; D006854:Hydrocortisone
J Renin Angiotensin Aldosterone Syst. 2008 Jun;9(2):103-6
18584586
Endocr J. 2007 Apr;54(2):287-93
17379961
Int J Urol. 2003 Oct;10(10):544-6
14516402
J Clin Endocrinol Metab. 2008 Sep;93(9):3266-81
18552288
Hypertens Res. 2006 Nov;29(11):883-9
17345788
Am J Hypertens. 2008 Nov;21(11):1269-72
18772856
Endocr J. 2001 Feb;48(1):103-11
11403095
Hypertens Res. 2003 Aug;26(8):663-8
14567506
Eur J Endocrinol. 2001 Mar;144(3):199-206
11248736
Clin Exp Pharmacol Physiol. 1995 Jun-Jul;22(6-7):460-2
8582103
Trends Endocrinol Metab. 2008 Apr;19(3):88-90
18314347
Am J Surg Pathol. 1989 Mar;13(3):202-6
2919718
Hypertens Res. 2004 Mar;27(3):193-202
15080378
Eur J Endocrinol. 2009 Mar;160(3):443-51
19131502
Am J Surg Pathol. 1984 Mar;8(3):163-9
6703192
Clin Endocrinol (Oxf). 1997 May;46(5):619-26
9231058
J Lab Clin Med. 1976 Aug;88(2):261-70
182890
Endocr J. 2009;56(3):495-502
19270420
Metabolism. 2002 Mar;51(3):350-5
11887172
Intern Med. 2008;47(1):37-42
18176003
J Clin Endocrinol Metab. 1969 Feb;29(2):239-50
4303579
Clin Exp Nephrol. 2008 Oct;12(5):382-387
18543063
Am J Med. 1972 Nov;53(5):627-37
4342887
J Clin Endocrinol Metab. 2003 May;88(5):1994-8
12727943
Hypertens Res. 2007 Apr;30(4):367-73
17541216
Hypertens Res. 2007 Nov;30(11):1083-95
18250558
J Clin Invest. 1973 Sep;52(9):2272-7
4353776
Horm Res. 1997;47(4-6):273-8
9167964
J Endocrinol. 1998 Mar;156(3):415-23
9582497
J Clin Endocrinol Metab. 1973 Nov;37(5):765-75
4356136
J Endocrinol. 2007 Oct;195(1):39-48
17911395
J Clin Endocrinol Metab. 1968 Jul;28(7):1006-13
4298942
J Clin Endocrinol Metab. 1976 Jul;43(1):115-31
947931
Clin Exp Nephrol. 2006 Jun;10(2):127-30
16791399
Clin Endocrinol (Oxf). 1974 Jan;3(1):41-8
4366076
Hypertens Res. 2008 Aug;31(8):1669-75
18971543
J Clin Endocrinol Metab. 1978 Apr;46(4):552-60
225341
Hypertens Res. 2001 Nov;24(6):723-6
11768734
Endocr J. 1994 Oct;41(5):471-82
7889106