Advertisement

Journal of Endocrinological Investigation

, Volume 23, Issue 6, pp 393–398 | Cite as

A giant prolactinoma presenting with unilateral exophthalmos: Effect of cabergoline and review of the literature

  • Joris Berwaerts
  • J. Verhelst
  • R. Abs
  • B. Appel
  • C. Mahler
Case Report

Abstract

We report the case of a 45-year-old male presenting with unilateral exophthalmos due to a large tumoral mass invading the skull base. Ophthalmologic examination did not show any visual field defects. Imaging techniques demonstrated extension of a huge tumor (approx. 8×8×8 cm) into the right orbit and nasopharynx. Endocrine work-up revealed grossly elevated serum prolactin (PRL) levels (26,466 μg/l, nl. <12), pointing to a large, invasive macroprolactinoma. Stimulation tests indicated associated partial adrenal and growth hormone deficiencies. Planned surgery was abandoned, and the patient was instead treated with the long-acting dopamine agonist, cabergoline. Over a period of one year, serum PRL dropped to 131 μg/l, while the tumor mass shrank to less than 50% of its original volume (with 3.5 mg/ week of cabergoline). The exophthalmos disappeared, and the patient did not develop rhinorrhea or any other side effects from treatment with cabergoline. The efficacy was maintained throughout the second year (ultimate serum PRL 74 μg/l, and final size less than 10% of the original). With reference to this case, we review other macroprolactinomas reported in the recent literature for associated exophthalmos, grossly elevated serum PRL levels (≥15,000 μg/l), and/or “giant” size (≥4 cm in maximum diameter). We highlight the use of dopamine agonists in the treatment of prolactinomas with such unusual characteristics.

Key-words

Prolactinoma giant exophthalmos hyperprolactinemia cabergoline 

Preview

Unable to display preview. Download preview PDF.

Unable to display preview. Download preview PDF.

References

  1. 1.
    Verhelst J., Abs R., Maiter D., van den Bruel A., Vandeweghe M., Velkeniers B., Mockel J., Lamberigts G., Petrossians P., Coremans P., Mahler C., Stevenaert A., Verlooy J., Raftopoulos C., Beckers A. Cabergoline in the treatment of hyperprolactinemia: a study in 455 patients. J. Clin. Endocrinol. Metab. 1999, 84: 2518–2522.PubMedCrossRefGoogle Scholar
  2. 2.
    Murphy F.Y., Vesely D.L., Jordan R.M., Flanigan S., Kohler P.O. Giant invasive prolactinomas. Am. J. Med. 1987, 83: 995–1002.PubMedCrossRefGoogle Scholar
  3. 3.
    Davis J.R.E., Sheppard M.C., Heath D.A. Giant invasive prolactinoma: a case report and review of nine further cases. Q. J. Med. 1990, 74: 227–238.PubMedGoogle Scholar
  4. 4.
    Grebe S.K.G., Delahunt J.W., Feek C.M. Treatment of extensively invasive (giant) prolactinomas with bromocriptine. N. Z. Med. J. 1992, 105: 129–131.PubMedGoogle Scholar
  5. 5.
    Kolodny J., Dluhy R.G. Recurrent prolactinoma and meningioma following irradiation and bromocriptine treatment. Am. J. Med. 1985, 78: 153–155.PubMedCrossRefGoogle Scholar
  6. 6.
    Ross R.J.M., McEniery J.M., Grossman A., Doniach I., Besser G.M., Savage M.O. Massive prolactinoma with galactorrhea in a prepubertal boy. Postgrad. Med. J. 1989, 65: 403–406.PubMedCentralPubMedCrossRefGoogle Scholar
  7. 7.
    Angyal E.A., Lee H-J., Wolansky L.J., Koenigsberger M.R., Nathanson D., Zimmer A.E. Prolactinoma invasion of superior ophthalmic vein: CT and MR findings. J. Comput. Assist. Tomogr. 1993, 17: 964–966.PubMedCrossRefGoogle Scholar
  8. 8.
    Levy R.A., Quint D.J. Giant pituitary adenoma with unusual orbital and skull base extension. Am. J. Rad. 1998, 170: 194–196.Google Scholar
  9. 9.
    Barkan A.L., Chandler W.F. Giant pituitary prolactinoma with falsely low serum prolactin: the pitfall of the “high-dose hook effect”, case report. Neurosurgery 1998, 42: 913–916.PubMedCrossRefGoogle Scholar
  10. 10.
    Daita G., Yonemasu Y., Hashizume A. Unilateral exophthalmos caused by an invasive pituitary adenoma. Neurosurgery 1987, 21: 716–718.PubMedCrossRefGoogle Scholar
  11. 11.
    Landolt A.M. Cerebrospinal fluid rhinorrhea: a complication of therapy for invasive prolactinomas. Neurosurgery 1982, 11: 395–401.PubMedCrossRefGoogle Scholar
  12. 12.
    Clayton R.N., Webb J., Heath D.A., Dunn P.J., Rolfe E.B., Hockley A.D. Dramatic and rapid shrinkage of a massive invasive prolactinoma with bromocriptine: a case report. Clin. Endocrinol. (Oxf.) 1985, 22: 573–581.CrossRefGoogle Scholar
  13. 13.
    Scherrer H., Turpin G., de Gennes J-L., Schaison-Cusin M., Heshmati H.M., Thibierge M., Metzger J. Résultats du traitement par bromocriptine des adénomes à prolactine géants ou expansifs. Ann. Méd. Interne (Paris) 1985, 136: 459–466.Google Scholar
  14. 14.
    Barrera C.M., Ruiz A., Banks W.A. A giant prolactinoma and the effect of chronic bromocriptine therapy on basal and TRH-stimulated serum prolactin levels. Horm. Res. 1991, 35: 167–169.PubMedCrossRefGoogle Scholar
  15. 15.
    van der Lely A-J., Knegt P.P.M., Stefanko S.Z., Tanghe H.L., Singh R., Lamberts S.W. Nasopharyngeal presentation of pituitary tumors: differential diagnosis and treatment. J. Clin. Endocrinol. Metab. 1992, 74: 811–813.PubMedCrossRefGoogle Scholar
  16. 16.
    Reuter U., Mehraein S., Arnold G., Lehmann R.A. A “giant” prolactinoma. J. Neurol. Neurosurg. Psychiatry. 1997, 63: 295.PubMedCentralPubMedCrossRefGoogle Scholar
  17. 17.
    Saeki N., Nakamura M., Sunami K., Yamaura A. Surgical indication after bromocriptine therapy on giant prolactinomas: effects and limitations of the medical treatment. Endocr. J. 1998, 45: 529–537.PubMedCrossRefGoogle Scholar
  18. 18.
    Cannavò S., Bartolone L., Blandino A., Spinella S., Galatioto S., Trimarchi F. Shrinkage of a PRL-secreting pituitary macroadenoma resistant to cabergoline. J. Endocrinol. Invest. 1999, 22: 306–309.PubMedGoogle Scholar
  19. 19.
    Symon L., Jakubowski J., Kendall B. Surgical treatment of giant pituitary adenomas. J. Neurol. Neurosurg. Psychiatry 1979, 42: 973–982.PubMedCentralPubMedCrossRefGoogle Scholar
  20. 20.
    Pia H.W., Grote E., Hildebrandt G. Giant pituitary adenomas. Neurosurg. Rev. 1985, 8: 207–220.PubMedCrossRefGoogle Scholar
  21. 21.
    Majós C., Coll S., Aguilera C., Acebes J.J., Pons L.C. Imaging of giant pituitary adenomas. Neuroradiology 1998, 40: 651–655.PubMedCrossRefGoogle Scholar
  22. 22.
    Perani D., Colombo N., Scotti G., Tonon C. Rapid size reduction of giant prolactinoma following medical treatment. J. Comput. Assist. Tomogr. 1984, 8: 131–133.PubMedCrossRefGoogle Scholar
  23. 23.
    Iwai Y., Hakuba A., Khosla V.K., Nishikawa M., Katsuyama J., Inoue Y., Nishimura S. Giant basal prolactinoma extending into the nasal cavity. Surg. Neurol. 1992, 37: 280–283.PubMedCrossRefGoogle Scholar
  24. 24.
    Cook R.J., Uttley D., Wilkins P.R., Archer D.J., Bell B.A. Prolactinomas in men masquerading as invasive skull base tumours. Br. J. Neurosurg. 1994, 8: 51–55.PubMedCrossRefGoogle Scholar
  25. 25.
    Cole D.R., Lees P.D., Armitage M. A good wife, but never a mother... J.R. Soc. Med. 1995, 88: 176P–178P.PubMedCentralPubMedGoogle Scholar
  26. 26.
    Biller B.M.K., Molitch M.E., Vance M.L., Baker Cannistraro K., Davis K.R., Simons J.A., Schoenfelder J.R., Klibanski A. Treatment of prolactin-secreting macroadenomas with the once-weekly dopamine agonist cabergoline. J. Clin. Endocrinol. Metab. 1996, 81: 2338–2343.PubMedGoogle Scholar
  27. 27.
    Holness R.O., Schlossberg A.H., Heffernan L.P.M. Cerebrospinal fluid rhinorrhea caused by bromocriptine therapy of prolactinoma. Neurology 1984, 34: 111–113.PubMedCrossRefGoogle Scholar
  28. 28.
    Hildebrandt G., Zierski J., Christophis P., Laun A., Schatz H., Lancranjan I., Klug N. Rhinorrhea following dopamine agonist therapy of invasive macroprolactinoma. Acta Neurochir. (Wien) 1989, 96: 107–113.CrossRefGoogle Scholar
  29. 29.
    Bronstein M.D., Musolino N.R., Benabou S., Marino R. Cerebrospinal fluid rhinorrhea occurring in long-term bromocriptine treatment of macro-prolactinomas. Surg. Neurol. 1989, 32: 246–249.CrossRefGoogle Scholar
  30. 30.
    Pascal-Vigneron V., Weryha G., Braun M., Morel-Jean J., Bisset S., Leclère J. La rhinorrhée et l’otorrhée: des complications rares du traitement médical des prolactinomes invasifs. Ann. Endocrinol. (Paris). 1993, 54: 347–351.Google Scholar
  31. 31.
    Ferrari C.I., Abs R., Bevan J.S., Brabant G., Ciccarelli E., Motta T., Mucci M., Muratori M., Musatti L., Verbessem G., Scanlon M.F. Treatment of macroprolactinoma with cabergoline: a study of 85 patients. Clin. Endocrinol. (Oxf.) 1997, 46: 409–413.CrossRefGoogle Scholar
  32. 32.
    Colao A., Di Sarno A., Landi M.L., Cirillo S., Sarnacchiaro F., Facciolli G., Pivonello R., Cataldi M., Merola B., Annunziato L., Lombardi G. Long-term and low-dose treatment with cabergoline induces macroprolactinoma shrinkage. J. Clin. Endocrinol. Metab. 1997, 82: 3574–3579.PubMedCrossRefGoogle Scholar
  33. 33.
    Bevan J.S., Webster J., Burke C.W., Scanlon M.F. Dopamine agonists and pituitary tumor shrinkage. Endocr. Rev. 1992, 13: 220–240.PubMedCrossRefGoogle Scholar
  34. 34.
    Cannavò S., Curtò L., Squadrito S., Almoto B., Vieni A., Trimarchi F. Cabergoline: a first-choice treatment in patient with previously untreated prolactin-secreting pituitary adenoma. J. Endocrinol. Invest. 1999, 22: 354–359.PubMedGoogle Scholar
  35. 35.
    Rains C.P., Bryson H.M., Fitton A. Cabergoline: a review of its pharmacological properties and therapeutic potential in the treatment of hyperprolactinaemia and inhibition of lactation. Drugs 1995, 49: 255–279.PubMedCrossRefGoogle Scholar

Copyright information

© Italian Society of Endocrinology (SIE) 2000

Authors and Affiliations

  • Joris Berwaerts
    • 2
  • J. Verhelst
    • 2
  • R. Abs
    • 1
  • B. Appel
    • 3
  • C. Mahler
    • 2
  1. 1.Department of NeuroradiologyMiddelheim HospitalBelgium
  2. 2.Department of EndocrinologyUniversity HospitalAntwerpBelgium
  3. 3.SmithKline Beecham ACCI - CRUAddenbrooke’s HospitalUK-CambridgeUK

Personalised recommendations