Multiple meningiomas in two male-to-female transsexual patients with hormone replacement therapy: A report of two cases and a brief literature review

Raj, Rahul; Korja, Miikka; Koroknay-Pál, Päivi; Niemelä, Mika

doi:10.4103/sni.sni_22_18

Cited by 10 publications

(12 citation statements)

References 18 publications

(26 reference statements)

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“…The majority (n = 10) were managed upfront with surgical intervention, and most also had CPA stopped at the same time (n = 8), except for one who had the dose tapered. 19 Two transgender persons were managed by cessation of CPA only without surgical intervention. 21,22 Synthesis of individual meningioma associated with included case reports Table 4 summarizes the individual meningioma (n = 35) described within the case reports included within this systematic review.…”

Section: Results Of Individual Case Reports and Associated Meningiomamentioning

confidence: 99%

“…As expected, all nine included studies were case reports from a hospital setting. One study reported on two individual patients, 19 while another reported on three individual patients. 20 Risk of bias within studies Risk of bias assessment was performed for each individual case report identified (n = 12).…”

Section: Study Characteristicsmentioning

confidence: 99%

“…Estradiol has been identified as an enhancer of meningioma cell proliferation in vivo, 26 although conflicting results exist as to whether there are increased meningioma growth rates for patients. 27,28 Reports in the literature also describe a preponderance of CPA-associated meningioma to the skull base, possibly explained by the differing embryonal origin of the skull base meninges, which raises the 36 1.2 R petrous n Millward et al 36 1.3 Olfactory groove y 2 Chordoid a GTR 15-60% + ve 90% + ve Millward et al 36 1.4 L greater sphenoid wing n Boer et al 21 2.1 R sphenoid n Boer et al 21 2.2 R cavernous sinus n Boer et al 21 2.3 Unknown (small) n Boer et al 21 2.4 Unknown (small) n Boer et al 21 2.5 Unknown (small) n Boer et al 21 2.6 Unknown (small) n Boer et al 21 2.7 Unknown (small) n Raj et al 19 3.1 Suprasellar y 1 Not stated GTR Lower High Raj et al 19 3.2 Cribriform plate y 1 Not stated GTR Lower High Raj et al 19 3.3 R frontal convexity (small) n Raj et al 19 3.4 R frontal convexity (large) y 1 Not stated GTR Lower High Raj et al 19 3.5 Suprasellar n Raj et al 19 4.1 L cavernous sinus y 1 Not stated STR Àve + ve Raj et al 19 4.2 L frontal convexity y 1 Not stated GTR Àve + ve Raj et al 19 4.3 L temporal convexity y 1 Not stated GTR Àve + ve Raj et al 19 4.4 R tentorial n Mancini et al 42 5.1 L occipital convexity y 1 Meningothelial GTR 20% + ve 90% + ve Ter Wengel et al 20 6.1 L ant clinoid y 2 Not stated GTR Àve Strongly + ve Ter Wengel et al 20 7.1 R tentorial y 1 Not stated GTR Weakly + ve + ve Ter Wengel et al 20 7.2 Unknown (small) n Ter Wengel et al 20 8.1 L sphenoid y 1 Not stated STR + ve Àve Ter Wengel et al 20 8.2 R frontal convexity n Ter Wengel et al 20 8.3 R frontal convexity n Ter Wengel et al 20 8.4 L frontal convexity n Ter Wengel et al 20 8.5 R parafalcine frontoparietal n Ter Wengel et al 20 8…”

Section: Findings From This Systematic Review Of Case Reportsmentioning

confidence: 99%

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Development and Growth of Intracranial Meningiomas in Transgender Women Taking Cyproterone Acetate as Gender-Affirming Progestogen Therapy: A Systematic Review

et al. 2022

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Background: Gender-affirming hormone therapy is critical to the management of transgender persons. Cyproterone acetate (CPA) is a synthetic, progesterone-like compound commonly used in high doses as gender-affirming progestogen therapy in transgender women. An association between high-dose CPA and the development and growth of intracranial meningioma, including case reports in transgender women, has been described. This systematic review summarizes these cases at the patient level and discusses their management. Methods: This systematic review was registered with PROSPERO (CRD42020191965). A detailed search of the PubMed, EMBASE, and Web of Science electronic bibliographic databases was performed (inception-December 20, 2020). Two review authors independently completed screening, data extraction, and risk of bias assessment in duplicate. Results: Nine records were included describing (n = 12) individual case reports and (n = 35) intracranial meningiomas. The median age at presentation was 48 years (interquartile range [IQR], 43-55 years), most frequent daily CPA doses were 50 mg/day (n = 5) and 100 mg/day (n = 5), and the median duration of CPA use was 9.5 years (IQR, 6.5-17.5 years). Multiple meningiomas were common (n = 7). For most cases (n = 10), surgical resection was the initial preferred management strategy, but two were successfully managed by CPA cessation. Conclusions: Transgender women receiving high doses of CPA may be at increased risk of intracranial meningioma development and/or growth, although this remains a rare disease. For presumed CPA-associated meningioma, drug cessation appears to be an appropriate management strategy when surgery is not imminently required to manage raised intracranial pressure or prevent neurological deterioration. Given the importance of gender-affirming hormone therapy to transgender persons, a suitable alternative hormone regimen should be offered, although the use of CPA in both high doses and for prolonged periods of time is now in decline.

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Section: Results Of Individual Case Reports and Associated Meningiomamentioning

confidence: 99%

Section: Study Characteristicsmentioning

confidence: 99%

Section: Findings From This Systematic Review Of Case Reportsmentioning

confidence: 99%

See 1 more Smart Citation

Development and Growth of Intracranial Meningiomas in Transgender Women Taking Cyproterone Acetate as Gender-Affirming Progestogen Therapy: A Systematic Review

et al. 2022

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“…Since numerous publications have reported cases of meningiomas in patients treated with CPA since 2007 [15,[19][20][21][22][23], our practitioners therefore systematically questioned patients about their current or past use of one or more hormonal treatments.…”

Section: Discussionmentioning

confidence: 99%

Meningiomas and cyproterone acetate: a retrospective, monocentric cohort of 388 patients treated by surgery or radiotherapy for intracranial meningioma

et al. 2021

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HAL is a multi-disciplinary open access archive for the deposit and dissemination of scientific research documents, whether they are published or not. The documents may come from teaching and research institutions in France or abroad, or from public or private research centers. L'archive ouverte pluridisciplinaire HAL, est destinée au dépôt et à la diffusion de documents scientifiques de niveau recherche, publiés ou non, émanant des établissements d'enseignement et de recherche français ou étrangers, des laboratoires publics ou privés.

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“…On the other hand, cyproterone acetate (not currently FDA approved in United States for use in patients with prostate cancer) is known to cross blood-brain barrier (Denmeade and Isaacs 2002). Cyproterone acetate is also known to worsen prolactinoma and hence has the potential to result in PA among patients with occult or known pituitary adenoma (Raj et al 2018). In our review, 3 patients with prior/concurrent use of cyproterone acetate developed PA.…”

Section: Author's Recommendationsmentioning

confidence: 95%

Pituitary apoplexy induced by gonadotropin-releasing hormone (GnRH) agonist administration for treatment of prostate cancer: a systematic review

Raj

Elshimy

Jacob

et al. 2021

J Cancer Res Clin Oncol

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Objective We aimed to review of literature on the clinical presentation, management and outcomes of pituitary apoplexy following gonadotrophic release hormone (GnRH) agonist administration for the treatment of prostate cancer. Methods We used PRISMA guidelines for our systematic review and included all English language original articles on pituitary apoplexy following GnRH agonist administration among prostate cancer patients from Jan 1, 1995 to Dec 31, 2020. Data on patient demographics, prostate cancer type, Gleason score at diagnosis, history of pituitary adenoma, clinical presentation, GnRH agonist, interval to pituitary apoplexy, laboratory evaluation at admission, radiologic findings, treatment of pituitary apoplexy, time to surgery if performed, pathology findings, and clinical/hormonal outcomes were collected and analyzed. Results Twenty-one patients with pituitary apoplexy met our inclusion criteria. The mean age of patients was 70 (60–83) years. Leuprolide was the most common used GnRH agonist, used in 61.9% of patients. Median duration to symptom onset was 5 h (few minutes to 6 months). Headache was reported by all patients followed by ophthalmoplegia (85.7%) and nausea/vomiting (71.4%). Three patients had blindness at presentation. Only 8 cases reported complete anterior pituitary hormone evaluation on presentation and the most common endocrine abnormality was FSH elevation. Tumor size was described only in 15 cases and the mean tumor size was 26.26 mm (18–48 mm). Suprasellar extension was the most common imaging finding seen in 7 patients. 71.4% of patients underwent pituitary surgery, while 23.8% were managed conservatively. Interval between symptoms onset to pituitary surgery was 7 days (1–90 days). Gonadotroph adenoma was most common histopathologic finding. Clinical resolution was comparable, while endocrine outcomes were variable among patients with conservative vs surgical management. Conclusion Although the use of GnRH agonists is relatively safe, it can rarely lead to pituitary apoplexy especially in patients with pre-existing pituitary adenoma. Physicians should be aware of this complication as it can be life threatening. A multidisciplinary team approach is recommended in treating individuals with pituitary apoplexy.

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Multiple meningiomas in two male-to-female transsexual patients with hormone replacement therapy: A report of two cases and a brief literature review

Cited by 10 publications

References 18 publications

Development and Growth of Intracranial Meningiomas in Transgender Women Taking Cyproterone Acetate as Gender-Affirming Progestogen Therapy: A Systematic Review

Development and Growth of Intracranial Meningiomas in Transgender Women Taking Cyproterone Acetate as Gender-Affirming Progestogen Therapy: A Systematic Review

Meningiomas and cyproterone acetate: a retrospective, monocentric cohort of 388 patients treated by surgery or radiotherapy for intracranial meningioma

Pituitary apoplexy induced by gonadotropin-releasing hormone (GnRH) agonist administration for treatment of prostate cancer: a systematic review

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