ESA-SRB-AOTA 2019

Cyproterone Related Meningioma In A Trans-sexual Female And Regression Following Its Discontinuation (#616)

Danish Mahmud 1 , Richard Gauci 1
  1. Fiona Stanley Hospital, Murdoch, WA, Australia

Introduction

Meningiomas are hormone sensitive tumours known to express progesterone, androgen and estrogen receptors (1). They are more common in women, particularly during pregnancy and reproductive years. Cyproterone acetate is used for androgen suppression in male to female transsexual patients, but also has anti-gonadotropic and progestational effects (2).  

Case Report

A 55-year-old male to female transsexual patient presented with impairment of vision in the left eye (temporal field defect and colour vision changes) after being on hormone management (Estradiol 12-18mg/day and Cyproterone acetate 100mg BD) therapy for twenty years, managed in the general practice setting. MRI showed multiple meningiomata with the left frontal meningioma measuring 9x13x11mm causing compression of the optic nerve. Additional lesions identified on MRI included a 10x10mm right petroclinoid meningioma, a 11x17mm left paraclinoid meningioma, and a 15x15mm right tentorial meningioma

Based on the symptomatic nature of left frontal meningioma, neurosurgical management was recommended which the patient declined. Cryproterone acetate was weaned and ultimately ceased and replaced with Spironolactone. Oestradiol dose was reduced, though negotiating a dose with the patient was difficult. Significant improvement in visual symptoms and reduction in size of meningiomata ensued over 12 months following discontinuation of Cyproterone acetate.

Discussion

Our case report adds to the previously established association linking long term high dose Cyproterone acetate (50mg/day) exposure to meningioma(3). There are also few case reports of regression in size of meningiomata with discontinuation of cyproterone acetate and oestradiol (4, 5). Our patient had significant clinical and radiological improvement with discontinuation of Cyproterone only (oestradiol was continued) avoiding the need for neurosurgery.

Key message:

A period of discontinuation of Cyproterone acetate may be reasonable first step in patients with symptomatic meningioma on long term Cyproterone acetate treatment.

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  3. Gil M, Oliva B, Timoner J, Maciá MA, Bryant V, de Abajo FJ. Risk of meningioma among users of high doses of cyproterone acetate as compared with the general population: evidence from a population-based cohort study. Br J Clin Pharmacol. 2011;72(6):965-8.
  4. Gonçalves AM, Page P, Domigo V, Méder JF, Oppenheim C. Abrupt regression of a meningioma after discontinuation of cyproterone treatment. AJNR Am J Neuroradiol. 2010;31(8):1504-5.
  5. Bernat AL, Bonnin S, Labidi M, Aldahak N, Bresson D, Bouazza S, et al. Regression of Giant Olfactory Groove Meningioma and Complete Visual Acuity Recovery after Discontinuation of Cyproterone Acetate. J Ophthalmic Vis Res. 2018;13(3):355-8.