Balcerek Matthew I, Hovelroud Rachel, Ruhl Matthew, Nolan Brendan J
Department of Endocrinology and Diabetes, Royal Brisbane and Women's Hospital, Herston, QLD 4029, Australia.
Faculty of Medicine, Royal Brisbane Clinical Unit, The University of Queensland, Herston, QLD 4029, Australia.
JCEM Case Rep. 2024 Oct 16;2(10):luae173. doi: 10.1210/jcemcr/luae173. eCollection 2024 Oct.
Progestin-associated meningioma is a rare complication of cyproterone acetate (CPA), an anti-androgen commonly prescribed in feminizing hormone therapy regimens for transgender and gender-diverse individuals. A dose-response association has been observed, particularly with longer-term exposure to doses ≥ 50 mg daily; however, the dose below which CPA use is safe remains unclear. We herein report the cases of 2 transgender women using CPA who developed meningioma. Novel aspects of our cases include: (i) the presence of symptomatic giant meningiomas (> 5 cm), including multiple meningiomas in one patient, requiring urgent surgical intervention; (ii) meningioma development with both high-dose, long duration and low-dose, shorter duration CPA; and (iii) the presence of a missense variant in one patient, which may play a role in the pathogenesis of progestin-associated meningioma. Our cases highlight the real-world risk of this likely underreported adverse effect and underscore the importance of clinician vigilance for neurological sequelae. We suggest using the lowest dose of CPA that maintains adequate androgen suppression, with consideration of alternative anti-androgens where appropriate.
孕激素相关的脑膜瘤是醋酸环丙孕酮（CPA）的一种罕见并发症，CPA是一种抗雄激素药物，常用于为跨性别和性别多样化个体制定的女性化激素治疗方案中。已观察到剂量反应关系，尤其是长期暴露于每日剂量≥50毫克的情况；然而，CPA使用安全的剂量下限仍不清楚。我们在此报告2例使用CPA的跨性别女性患脑膜瘤的病例。我们病例的新特点包括：（i）存在有症状的巨大脑膜瘤（>5厘米），其中1例患者有多个脑膜瘤，需要紧急手术干预；（ii）高剂量、长时间和低剂量、短时间使用CPA均出现脑膜瘤；（iii）1例患者存在错义变异，这可能在孕激素相关脑膜瘤的发病机制中起作用。我们的病例突出了这种可能未得到充分报告的不良反应在现实世界中的风险，并强调了临床医生对神经后遗症保持警惕的重要性。我们建议使用维持足够雄激素抑制的最低剂量的CPA，并在适当情况下考虑使用替代抗雄激素药物。
