Skip to main content

Subclinical Cushing’s syndrome associated with an adrenocortical oncocytoma

Journal of Endocrinological Investigation volume 31, pages 675–679 (2008)Cite this article

Abstract

Oncocytoma is a neoplasm that can arise in several organs, and it has been more commonly described in the kidney, salivary gland and thyroid. Oncocytoma arising in the adrenal gland is a rare finding. Moreover, functioning adrenocortical oncocytoma is exceptionally rare. A 47-yr-old man was incidentally discovered to have a right adrenal mass. The patient had no clinical features suggestive of increased adrenal function. However, hormonal evaluation showed a disturbed cortisol circadian rhythm, supranormal urinary cortisol excretion, a low level of ACTH, and a lack of suppressibility of cortisol secretion after dexamethasone. Right adrenalectomy was performed, and this revealed a well-circumscribed dark-brown tumor that measured 2.4x2.2 cm. The tumor consisted almost exclusively of large eosinophilic and epitheloid cells whose cytoplasm was packed with eosinophilic granulations, which corresponded to the numerous mitochondria confirmed on electron microscopy. This is a rare case of subclinical Cushing’s syndrome that was caused by adrenocortical oncocytoma.

This is a preview of subscription content, access via your institution.

Access options

Buy single article

Instant access to the full article PDF.

43,34 €

Price includes VAT (Finland)
Tax calculation will be finalised during checkout.

References

  1. Tallini G. Oncocytic tumours. Virchows Arch 1998, 433: 5–12.

    PubMed  Article  CAS  Google Scholar 

  2. Kakimoto S, Yushita Y, Sanefuji T, et al. Non-hormonal adrenocortical adenoma with oncocytoma-like appearances. Hinyokika Kiyo 1986, 32: 757–63.

    PubMed  CAS  Google Scholar 

  3. el-Naggar AK, Evans DB, Mackay B. Oncocytic adrenal cortical carcinoma. Ultrastruct Pathol 1991, 15: 549–56.

    PubMed  Article  CAS  Google Scholar 

  4. Gandras EJ, Schwartz LH, Panicek DM, Levi G. Case report. Adrenocortical oncocytoma: CT and MRI findings. J Comput Assist Tomogr 1996, 20: 407–9.

    PubMed  Article  CAS  Google Scholar 

  5. Sasano H, Suzuki T, Sano T, Kameya T, Sasano N, Nagura H. Adrenocortical oncocytoma. A true nonfunctioning adrenocortical tumor. Am J Surg Pathol 1991, 15: 949–56.

    Article  CAS  Google Scholar 

  6. Waters PR, Haselhuhn GD, Gunning WT, Phillips ER, Selman SH. Adrenocortical oncocytoma: two case reports and review of literature. Urology 1997, 49: 624–8.

    PubMed  Article  CAS  Google Scholar 

  7. Erlandson RA, Reuter VE. Oncocytic adrenal cortical adenoma. Ultrastruct Pathol 1991, 15: 539–47.

    PubMed  Article  CAS  Google Scholar 

  8. Begin LR. Adrenocortical oncocytoma: case report with immunocytochemical and ultrastructural study. Virchows Arch A Pathol Anat Histopathol 1992, 421: 533–7.

    PubMed  Article  CAS  Google Scholar 

  9. Lin BT, Bonsib SM, Mierau GW, Weiss LM, LJ Medeiros LJ. Oncocytic adrenocortical neoplasms: a report of seven cases and review of the literature. Am J Surg Pathol 1998, 22: 603–14.

    PubMed  Article  CAS  Google Scholar 

  10. Nguyen GK, Vriend R, Ronaghan D, Lakey WH. Heterotopic adrenocortical oncocytoma. A case report with light and electron microscopic studies. Cancer 1992, 70: 2681–4.

    CAS  Google Scholar 

  11. Macchi C, Rebuffat P, Blandamura S, et al. Adrenocortical oncocytoma: case report and review of the literature. Tumori 1998, 84: 403–7.

    PubMed  CAS  Google Scholar 

  12. Vara Castrodeza A, Ablanedo Ablanedo P, García García J, Martinez Lara CM, Díaz A, Calvo Blanco J. Adrenal oncocytoma. Arch Esp Urol 1999, 52: 525–8.

    PubMed  CAS  Google Scholar 

  13. Kitching PA, Patel V, Harach HR. Adrenocortical oncocytoma. J Clin Pathol 1999, 52: 151–3.

    PubMed Central  PubMed  Article  CAS  Google Scholar 

  14. Bisceglia M, Ludovico O, Di Mattia A, et al. Adrenocortical oncocytic tumors: report of 10 cases and review of the literature. Int J Surg Pathol 2004, 12: 231–43.

    PubMed  Article  Google Scholar 

  15. Poretti D, Mazzarol G, Bonomo G, Casadio C, Bellomi M. Adrenocortical oncocytoma: case report. Clin Imaging 2003, 27: 426–30.

    PubMed  Article  Google Scholar 

  16. Lee R, Al-Ahmadie HA, Boorjian SA, et al. A case of incidental adrenocortical oncocytoma. Nat Clin Pract Urol 2006, 11: 618–21.

    Article  Google Scholar 

  17. Xiao GQ, Pertsemlidis DS, Unger PD. Functioning adrenocortical oncocytoma: a case report and review of the literature. Ann Diagn Pathol 2005, 9: 295–7.

    PubMed  Article  Google Scholar 

  18. Segal S, Cytron S, Shenhav S, Gemer O. Adrenocortical oncocytoma in pregnancy. Obstet Gynecol 2001, 98: 916–8.

    PubMed  Article  CAS  Google Scholar 

  19. Gumy-Pause F, Bongiovanni M, Wildhaber B, Jenkins JJ, Chardot C, Ozsahin H. Adrenocortical oncocytoma in a child. Pediatr Blood Cancer 2008, 50: 718–21.

    PubMed  Article  Google Scholar 

  20. Song SY, Park S, Kim SR, Suh YL. Oncocytic adrenocortical carcinomas: a pathological and immunohistochemical study of four cases in comparison with conventional adrenocortical carcinomas. Pathol Int 2004, 54: 603–10.

    PubMed  Article  Google Scholar 

  21. Gabrilove JL, Sharma DC, Wotiz HH, Dorfman RI. Feminizing adrenocortical tumors in the male. A review of 52 cases including a case report. Medicine (Baltimore) 1965, 44: 37–79.

    CAS  Google Scholar 

  22. Bertagna C, Orth DN. Clinical and laboratory findings and results of therapy in 58 patients with adrenocortical tumors admitted to a single medical center (1951 to 1978). Am J Med 1981, 71: 855–67.

    PubMed  Article  CAS  Google Scholar 

  23. Veldhuis JD, Sowers JR, Rogol AD, Klein FA, Miller N, Dufau ML. Pathophysiology of male hypogonadism associated with endogenous hyperestrogenism. Evidence for dual defects in the gonadal axis. N Engl J Med 1985, 312: 1371–5.

    CAS  Google Scholar 

  24. Young J, Bulun SE, Agarwal V, et al. Aromatase expression in a feminizing adrenocortical tumor. J Clin Endocrinol Metab 1996, 81: 3173–6.

    PubMed  CAS  Google Scholar 

  25. Kuhn JM, Lefebvre H, Duparc C, Pellerin A, Luton JP, Strauch G. Cosecretion of estrogen and Inhibin B by a feminizing adrenocortical adenoma: Impact on gonadotropin secretion. J Clin Endocrinol Metab 2002, 87: 2367–75.

    PubMed  Article  CAS  Google Scholar 

  26. Bouraïma H, Lireux B, Mittre H, et al. Major hyperestrogenism in a feminizing adrenocortical adenoma despite a moderate overexpression of the aromatase enzyme. Eur J Endocrinol 2003, 148: 457–61.

    PubMed  Article  Google Scholar 

  27. Chang A, Harawi SJ. Oncocytes, oncocytosis, and oncocytic tumors. Pathol Annu 1992, 27: 263–304.

    PubMed  Google Scholar 

  28. Mantero F, Terzolo M, Arnaldi G, et al. A survey on adrenal incidentaloma in Italy. Study Group on Adrenal Tumors of the Italian Society of Endocrinology. J Clin Endocrinol Metab 2000, 85: 637–44.

    PubMed  CAS  Google Scholar 

  29. Terzolo M, Osella G, Alì A, et al. Subclinical Cushing’s syndrome in adrenal incidentaloma. Clin Endocrinol 1998, 48: 89–97.

    Article  CAS  Google Scholar 

  30. Weiss LM, Medeiros LJ, Vickery AL Jr. Pathologic features of prognostic significance in adrenocortical carcinoma. Am J Surg Pathol 1989, 13: 202–6.

    PubMed  Article  CAS  Google Scholar 

  31. Aubert S, Wacrenier A, Leroy X, et al. Weiss system revisited: a clinicopathologic and immunohistochemical study of 49 adrenocortical tumors. Am J Surg Pathol 2002, 26: 1612–9.

    PubMed  Article  Google Scholar 

  32. Cho EY, Ahn GH. Immunoexpression of inhibin alpha-subunit in adrenal neoplasms. Appl Immunohistochem Mol Morphol 2001, 9: 222–8.

    PubMed  Article  CAS  Google Scholar 

  33. Alsabeh R, Mazoujian G, Goates J, Medeiros LJ, Weiss LM. Adrenal cortical tumors clinically mimicking pheochromocytoma. Am J Clin Pathol 1995, 104: 382–90.

    PubMed  CAS  Google Scholar 

Download references

Author information

Affiliations

  1. Division of Endocrinology and Metabolism, Department of Internal Medicine, St. Mary’s Hospital, The Catholic University of Korea, # 62 Yoido-dong, Youngdeungpo-gu, Seoul, 150-713, Korea

    S. S. Lee, K. H. Baek MD, J. M. Lee, M. I. Kang, B. Y. Cha, K. W. Lee, H. Y. Son & S. K. Kang

  2. Department of Clinical Pathology, The Catholic, University of Korea, College of Medicine, Seoul, Korea

    Y. S. Lee

Authors
  1. S. S. Lee
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. K. H. Baek MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Y. S. Lee
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. J. M. Lee
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. M. I. Kang
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. B. Y. Cha
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. K. W. Lee
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. H. Y. Son
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. S. K. Kang
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to K. H. Baek MD.

Rights and permissions

Reprints and Permissions

About this article

Cite this article

Lee, S.S., Baek, K.H., Lee, Y.S. et al. Subclinical Cushing’s syndrome associated with an adrenocortical oncocytoma. J Endocrinol Invest 31, 675–679 (2008). https://doi.org/10.1007/BF03345624

Download citation

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/BF03345624

Key-words

  • Adrenal gland
  • subclinical Cushing’s syndrome
  • oncocytoma