Skip to main content
SpringerLink
Log in

Coexistence of unilateral adrenal macronodule and Cushing’s disease. Report of two cases

  • Case Report
  • Published:
Journal of Endocrinological Investigation Aims and scope Submit manuscript

Abstract

The coexistence of an unilateral adrenal macronodule and Cushing’s disease is well documented in only few reports. We hereby describe two patients with ACTH-dependent Cushing’s syndrome and asymmetric adrenal involvement. In both patients the left-sided adrenal contained a nodule sized 1.5 and 1.4 cm, respectively, while the contralateral gland appeared as normal at abdominal computerized tomography. Adrenal scintiscan revealed a bilateral uptake of the tracer. Dynamic testing of hypothalamic-pituitary-adrenal axis was suggestive of pituitary dependency but neuroradiologic imaging was inconclusive. Inferior petrosal sinus sampling demonstrated a clear center to periphery ACTH gradient and an ACTH-producing pituitary microadenoma was found and removed in both cases. Hypopituitarism ensued postoperatively and Cushing’s syndrome resolved but the adrenal nodules regressed only many month after operation. The present cases confirm that the presence of an adrenal mass in a patient with Cushing’s syndrome is insufficient to confidently conclude for the adrenal dependency of the syndrome.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  1. Findling J.W., Doppman J.L. Biochemical and radiologic diagnosis of Cushing’s syndrome. Endocrinol. Metab. Clin. North Am. 23: 511, 1994.

    PubMed  CAS  Google Scholar 

  2. Doppman J.L, Miller D.L., Dwyer A.J. Macronodular adrenal hyperplasia in Cushing disease. Radiology 166: 345, 1988.

    Google Scholar 

  3. Samuels M.H., Loriaux D.L. Cushing’s syndrome and the nodular adrenal gland. Endocrinol. Metab. Clin. North Am. 23: 555, 1994.

    PubMed  CAS  Google Scholar 

  4. Schteingart D.E., Tsao H.S. Coexistence of pituitary adrenocorticotropin-dependent Cushing’s syndrome with a solitary adrenal adenoma. J. Clin. Endocrinol. Metab. 50: 961, 1980.

    Article  PubMed  CAS  Google Scholar 

  5. Aron D.C., Findling J.W., Fizgerald P.A., Brooks R.M., Fisher F.E., Forsham P.H., Tyrrel J.B. Pituitary ACTH dependency of nodular adrenal hyperplasia in Cushing’s syndrome. Am. J. Med. 71: 302, 1981.

    Article  PubMed  CAS  Google Scholar 

  6. Lamberts S.W.J., Bons E.G., Bruining H.A. Different sensitivity to adrenocorticotropin of dispersed adrenocortical cells from patient with Cushing’s disease with macronodular and diffuse adrenal hyperplasia. J. Clin. Endocrinol. Metab. 58: 1106, 1984.

    Article  PubMed  CAS  Google Scholar 

  7. Leiba S., Shindel B., Weinberger I., Fuchs J. Rotenberg Z., Mor C., Kaufman H. Cushing’s disease coexisting with a single macronodule stimulating adenoma of the adrenal cortex. Acta Endocrinol. (Copenh.) 112: 323, 1986.

    CAS  Google Scholar 

  8. Hermus A.R., Pieters G.F., Smals A.G., Pesman G.J., Lamberts S.W. Benraad T.J. van Haelst U.J., Kloppenborg P.W. Transition from pituitary — dependent to adrenal — dependent Cushing’s syndrome. N. Engl. J. Med. 318: 966, 1988.

    Article  PubMed  CAS  Google Scholar 

  9. Nobels F., Abs R., Beckers A. Coexsistence of Cushing’s disease with unilateral uninodular hyperplasia. J. Endocrinol. Invest. 14(Suppl. 1): 88, 1991.

    Google Scholar 

  10. Fish H.R., Sobel D.O. Macronodular adrenal hyperplasia with hypothalamic — pituitary — adrenal suppression by ultra — high dose dexamethasone: regression following hypophysectomy. Clin. Neuropharm 9: 303, 1986.

    Article  CAS  Google Scholar 

  11. Watanobe H., Kawagishi T., Hirai Y. Cushing’s syndrome presenting the coexistence of a pituitary corticotrophic cell hyperplasia and a unilateral functional adrenal adenoma. Acta Endocrinol. (Copenh.) 110: 302, 1985.

    CAS  Google Scholar 

  12. Smals A.G.H., Pieters G.F.F.M., van Haelst U.J.G., Kloppenborg P.W.C. Macronodular Adrenocortical Hyperplasia in longstanding Cushing’ Disease. J. Clin. Endocrinol. Metab. 58: 25, 1984.

    Article  PubMed  CAS  Google Scholar 

  13. Hocher B., Bahr V. Hypercorticolism with non — pigmented micronodular adrenal hyperplasia: transition from pituitary — dependent to adrenal Cushing’s syndrome. Acta Endocrinol. (Copenh.) 128: 120, 1993.

    CAS  Google Scholar 

  14. Jackson J.A., Fachnie J.D., Mellinger R.C. Marked elevation of serum dehydroepiandrosterone sulphate in Cushing’s disease with macronodular adrenocortical hyperplasia. J. Endocrinol. Invest. 12: 269, 1989.

    Article  PubMed  CAS  Google Scholar 

  15. Findlay J.C., Sheeeler L.R., Engeland W.C., Aron D.C. Familial adrenocorticotropin-indipendent Cushing’s syndrome with bilateral macronodular adrenal hyperplasia. J. Clin. Endocrinol. Matab. 76: 189, 1993.

    CAS  Google Scholar 

  16. Gross M.D., Shapiro B., Francis I.R., Glazer G.M., Bree L.R., Arcomano MA, Schteingart D.E., McLeod M.K., Sanfield J.A, Thompson N.W. Scintigraphic evaluation of clinically silet adrenal masses. J. Nucl. Med. 35: 1145, 1994.

    PubMed  CAS  Google Scholar 

  17. Hedeland H., Ostberg G., Hokfelt B. On the prevalence of adrenocortical adenomas in an autopsy material in relation to hypertension and diabetes. Acta Med. Scand. 184: 211, 1967.

    Article  Google Scholar 

  18. Bunch F.T., Warner B.A. Cushing’s disease with a unilateral adrenal macronodule: evolutive transition or incidental finding? South Med. J. 86: 235, 1993.

    Article  PubMed  CAS  Google Scholar 

  19. Glazer H.S., Weyman P.J., Sagel S.S., Levitt R.G., McClennan B.L. Nonfunctioning adrenal masses: incidental discovery on computed tomography. Am. J. Roentgenol 139: 81, 1982.

    Article  CAS  Google Scholar 

  20. Herrera M.F., Grant C.S., Van Heerden J.A., Sheedy II P.F., IIstrup D.M. Incidentally discovered adrenal tumors: an institutional perspective. Surgery 110: 1014, 1991.

    PubMed  CAS  Google Scholar 

  21. Kaye T.B., Crapo L. The Cushing’s Syndrome: an update on diagnostic tests. Ann. Intern. Med. 112: 434, 1990.

    Article  PubMed  CAS  Google Scholar 

  22. O’Brien T., Young W.F., Davila D.G., Scheithaur B.W., Kovacs K., Horvath E., Vale W., van Heerden J.A. Cushing’s syndrome associated with ectopic production of corticotrophin-releasing hormone, corticotropin and vasopression by a pheochromocytoma. Clin Endocrinol. (Oxf.) 37: 460, 1992.

    Article  Google Scholar 

  23. Terzolo M., Alì A., Pisa A., Bollito E., Reimondo G., Paccotti P., Scardapane R., Angeli A. Cyclic Cushing’s syndrome due to ectopic secretion by an adrenal pheochromocytoma. J. Endocrinol. Invest. 17: 869, 1994.

    Article  PubMed  CAS  Google Scholar 

  24. Kelly W.F., Barnes A.J., Cassar J. Cushing’s syndrome due to adrenocortical carcinoma. A comprehensive clinical and biochemical study of patients treated by surgery and chemotherapy. Acta Endocrinol. (Copenh.) 91: 303, 1979.

    CAS  Google Scholar 

  25. King D.R., Lack E.F. Adrenal cortical carcinoma. A clinical and pathologic study of 49 cases. Cancer 44: 239, 1979.

    CAS  Google Scholar 

  26. Howlett A., Grossman A., Rees L.H., Besser G.M. Differential diagnosis on Cushing’s syndrome. Lancet 2: 87, 1986.

    Google Scholar 

  27. Fig L.M., Gross M.D., Shapiro B. Ehrmann D.A., Freitas J.E., Schteingart D.E., Glazer G.M., Francis I.R. Adrenal localization in the adrenocoticotropic hormone-independent Cushing syndrome. Ann. Intern. Med. 109: 547, 1988.

    Article  PubMed  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Divisione di Endocrinologia, Ospedale S. Croce, Via M. Coppino 26, 12100 Cuneo, Italy

    G. Borretta M.D., F. Cesario & I. Meineri

  2. Dipartimento di Scienze Cliniche e Biologiche, Cattedra di Medicina Interna, Ospedale S. Luigi, Università di Torino, Italy

    M. Terzolo, A. Pia & A. Angeli

Authors
  1. G. Borretta M.D.
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. M. Terzolo
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. F. Cesario
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. I. Meineri
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. A. Pia
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. A. Angeli
    View author publications

    You can also search for this author in PubMed Google Scholar

Rights and permissions

Reprints and permissions

About this article

Cite this article

Borretta, G., Terzolo, M., Cesario, F. et al. Coexistence of unilateral adrenal macronodule and Cushing’s disease. Report of two cases. J Endocrinol Invest 19, 131–135 (1996). https://doi.org/10.1007/BF03349849

Download citation

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/BF03349849

Key-words

Search

Navigation