Abstract
With increasing case reports, it has been indicated that some cases with adrenocorticotropic hormone (ACTH)-independent macronodular adrenal hyperplasia (AIMAH) show abnormal responses in cortisol to various stimulation tests. Here we report a case of AIMAH that showed an aberrant response to angiotensin II via AT1 receptor in cortisol hypersecretion. A 53-yr-old man was admitted to our division seeking further examinations for the possible diagnosis of Cushing's syndrome. He had hypertension, diabetes mellitus, and physical stigmata, such as moon face and central obesity. His plasma ACTH level was undetectable, and plasma cortisol level was high. Plasma cortisol showed no normal diurnal rhythm and was not suppressed after the administration of 8 mg of dexamethasone. Abdominal computed tomography demonstrated nodular enlargement of bilateral adrenal glands. He was diagnosed with Cushing's syndrome owing to AIMAH. An injection of arginine vasopressin (AVP) increased plasma cortisol and aldosterone levels, whereas ACTH remained undetectable. After 4 h in an upright position, plasma cortisol and aldosterone levels were increased. Pretreatment with candesartan, angiotensin II receptor AT1 antagonist, blocked the increase in plasma cortisol level. These results suggested a possibility of adrenal hypersensitivity to angiotensin II and AVP in cortisol secretion. Bilateral laparoscopic adrenalectomy was performed. The histological findings of the specimen were compatible with AIMAH. In summary, we have made the first report on a case of AIMAH with possible hypersensitivity to angiotensin II.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
References
Orth, D. N. and Kovacs, W. J. (1998). In: Williams textbook of endocrinology. Wilson, J. D., Foster, D. W., Kronenberg, H. M., and Larsen, P. R. (eds.) W. B. Saunders Philadelphia.
Cheitillin, R. A., Westphal, M., Cabrera, C. M., Fujii, D. K., Snyder, J., and Fitzgerald, P. A. (1988). Horm. Res. 29, 162–167.
Malchoff, C. D., Rosa, J., DeBold, C. R., Kozol, R. A., Ramsby, G. R., Page, D. L., Malchoff, D. M., and Orth, D. N. (1989). J. Clin. Endocrinol. Metab. 68, 855–860.
Lacroix, A., N'Diaye, N., Mircescu, H., Hamet, P., and Tremblay, J. (1998). Endocr. Res. 24, 835–843.
Horiba, N., Suda, T., Aiba, M., Naruse, M., Nomura, K., Imamura, M., and Demura, H. (1995). J. Clin. Endocrinol. Metab. 80, 2336–2341.
Iida, K., Kaji, H., Matsumoto, H., Okimura, Y., Abe, H., Fujisawa, M., Kamidono, S., and Chihara, K. (1997). Clin. Endocrinol. (Oxf.) 47, 739–745.
Daidoh, H., Morita, H., Hanafusa, J., Mune, T., Murase, H., Sato, M., Shibata, T., Suwa, T., Ishizuka, T., and Yasuda, K. (1998). Clin. Endocrinol. (Oxf.) 48, 403–409.
Lacroix, A., Tremblay, J., Touyz, R. M., Deng, L. Y., Lariviere, R., Cusson, J. R., Schiffrin, E. L., and Hamet, P. (1997). J. Clin. Endocrinol. Metab. 82, 2414–2422.
Lacroix, A., Tremblay, J., Rousseau, G., Bouvier, M., and Hamet, P. (1997). N. Engl. J. Med. 337, 1429–1434.
Lacroix, A., Hamet, P., and Boutin, J. M. (1999). N. Engl. J. Med. 341, 1577–1581.
Lacroix, A., Bolté, E., Tremblay, J., Dupre, J., Poitras, P., Fournier H., Garon, J., Garrel, D., Bayard, F., Taillefer, R., Flanagan, R. J., and Hamet, P. (1992). N. Engl. J. Med. 327, 974–980.
Reznik, Y., Allali-Zerah, V., Chayvialle, J. A., Leroyer, R., Leymarie, P., Travert, G., Lebrethon, M. C., Budi, I., Balliere, A. M., and Mahoudeau, J. (1992). N. Engl. J. Med. 327, 981–986.
Pralong, F. P., Gomez, F., Guillou, L., Mosimann, F., Franscella, S., and Gaillard, R. C. (1999). J. Clin. Endocrinol. Metab. 84, 3817–3822.
Aron, D. C., Findling, J. W., Fitzgerald, P. A., Brooks, R. M., Fisher, F. E., Forsham, P. H., and Tyrrell, J. B. (1981). Am. J. Med. 71, 302–306.
Hermus, A. R., Pieters, G. F., Smals, A. G., Pesman, G. J., Lamberts, S. W., Benraad, T. J., van Haelst, U. J., and Kloppenborg, P. W. (1988). N. Engl. J. Med. 318, 966–971.
Sasano, H., Suzuki, T., and Nagura, H. (1994). Mod. Pathol. 7, 215–219.
Perraudin, V., Delarue, C., Lefebvre, H., Contesse, V., Kuhn, J.-M., and Vaudry, H. (1993). J. Clin. Endocrinol. Metab. 76, 1522–1528.
Pralong, F. P., Roduit, R., Waeber, G., Castillo, E., Mosimann, F., Thorens, B., and Gaillard, R. C. (1998). Endocrinology 139, 4264–4268.
Dluhy, R. G. and Williams, G. H., In: Williams textbook of endocrinology. Wilson, J. D., Foster, D. W., Kronenberg, H. M., and Larsen, P. R. (eds.). W. B. Saunders: Philadelphia.
Lumbers, E. R. (1999). Regul. Pept. 80, 91–100.
Guillon, G., Trueba, M., Joubert, D., Grazzini, E., Chouinard, L., Côté, M., Payet, M. D., Manzoni, O., Barberis, C., Robert, M., and Gallo-Payet, N. (1995). Endocrinology 136, 1285–1295.
Tanabe, A., Naruse, M., Arai, K., Naruse, K., Yoshimoto, T., Seki, T., Imai, T., Miyazaki, H., Zeng, Z. P., Demura, R., and Demura, H. (1998). Horm. Metab. Res. 30, 490–495.
Mircescu, H., Jilwan, J., N'Diaye, N., Bourdeau, I., Tremblay, J., Hamet, P., and Lacroix, A. (2000). J. Clin. Endocrinol. Metab. 85, 3531–3536.
Iwasaki, Y., Oiso, Y., Yamauchi, K., Takatsuki, K., Kondo, K., Hasegawa, H., Itatsu, T., Niinomi, M., and Tomita, A. (1989). J. Clin. Endocrinol. Metab. 68, 560–565.
Gill, G. N., Ill, C. R., and Simonian, M. H. (1977). Proc. Natl. Acad. Sci. USA 74, 5569–5573.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Nakamura, Y., Son, Y., Kohno, Y. et al. Case of adrenocorticotropic hormone-independent macronodular adrenal hyperplasia with possible adrenal hypersensitivity to angiotensin II. Endocr 15, 57–61 (2001). https://doi.org/10.1385/ENDO:15:1:057
Received:
Revised:
Accepted:
Issue Date:
DOI: https://doi.org/10.1385/ENDO:15:1:057