Regression of giant olfactory groove meningioma and complete visual acuity recovery after discontinuation of cyproterone acetate

Bernat, Anne Laure; Bonnin, Sophie; Labidi, Moujahed; Aldahak, Nouman; Bresson, D.; Bouazza, S.; Froelich, Sébastien

doi:10.4103/jovr.jovr_21_17

Cited by 21 publications

(8 citation statements)

References 8 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…A strong association between cyproterone acetate (CA), a progesterone derivate and meningioma was rst reported in 2008 by Froelich et al [10], in a series of 9 patients with tumor regression after CA discontinuation. Since then, multiple similar reports have been published, and a clear relationship between meningioma and CA is now well established [11][12][13][14][15]. A recent study, based on the French national SNDS database, showed that women who initiated high dose CA treatment had a sevenfold increased risk of surgically treated meningioma with a strong dose-effect.…”

Section: Introductionmentioning

confidence: 93%

WITHDRAWN: Nomegestrol Acetate or Chlormadinone Acetate Progestative Treatment in Women: Meningioma Behavior at Treatment Discontinuation

Florea

Boissonneau

Passeri

et al. 2021

Preprint

Self Cite

View full text Add to dashboard Cite

Background: Associations between progestins and meningiomas is now well established. While the link between cyproterone acetate (CA) and meningioma was thoroughly studied, there is far less available data regarding the link between chlormadinone acetate (CHA) or nomegestrol acetate (NA) and risk of intracranial meningiomaMethods: We are presenting a series of 28 patients diagnosed with single or multiple meningiomas while treated with CHA-NA, in which the clinical and radiological course were analyzed after treatment discontinuation.Results: 28 women, with a mean age of 56 years old, were diagnosed with one or multiple meningioma while being treated with either CHA or NA. After stopping treatment, 89.3% showed either tumor shrinkage or tumor stabilization on follow-up MRIs. Multiple meningiomas were more likely observed in patients with long periods of treatment (>10 years, p 0.03) and seem to have a better clinical course (p 0.01). Most of the lesions were located on the skull base (55.4%). Four patients with multiple meningiomas showed discordant tumors evolution, with some tumors growing while others were decreasing. Most of the growing meningiomas were either convexity or midline lesions and more posteriorly located. Conclusion: Our study demonstrated a significant percentage of tumor diminution or stabilization after NA and CHA discontinuation. Therefore, treatment discontinuation with close monitoring should be the first measure taken if urgent surgery is not indicated. However, our results seem to be less encouraging than previously described in patients treated by CA, with more patients showing tumor growth despite treatment discontinuation. Further studies are needed to differentiate the effect of the different progestins treatment on meningiomas.

show abstract

Section: Introductionmentioning

confidence: 93%

WITHDRAWN: Nomegestrol Acetate or Chlormadinone Acetate Progestative Treatment in Women: Meningioma Behavior at Treatment Discontinuation

Florea

Boissonneau

Passeri

et al. 2021

Preprint

Self Cite

View full text Add to dashboard Cite

show abstract

“…The suspicion of a link between CPA and meningiomaswas raised by our group in 2008[6]and was con rmedby numerous case seriesshowing regression or stabilization of meningioma(s) after CPA discontinuation [7,8,12,13,15,17,18,27,[29][30][31][32]. Gill et al [14]described an increased risk of meningioma(s)in patients under high dose CPA and with a longer than 1 year exposure.…”

Section: Discussionmentioning

confidence: 99%

“…Since 2009, following the results of our CPA studies [7,8,12,13,20,27], every patient presenting with a meningioma to our neurosurgical department was questioned about previous history of progestin treatment.Every newly diagnosed patient with intracranial tumor radiologically compatible with a meningioma associated with CPA intake, followed aconservative strategy starting with discontinuation of CPA and a close clinical and radiological follow-up.Surgery was onlyindicated in case of tumor growthon serial magnetic resonance imaging (MRI)or in severely symptomatic patients.…”

Section: Patients' Selectionmentioning

confidence: 99%

“…Cyproterone acetate (CPA) is a synthetic progestin drug with strong antiandrogenic effect used for a variety of indications, mostly related to hyperandrogenization and hirsutism in women, while it is used for prostate cancer and paraphilia in men.Thesuspicion of arelationship between the use of CPA and the risk of intracranial meningiomasand, the regression after CPA discontinuation was rst reported in 2008 by Froelich et al[6] and was followed by numerous case reports or series [7][8][9][10][11][12][13][14][15][16][17][18].This increased risk and a strong dose-effect wererecently proven in a large epidemiological study in France [19]. For progestinrelated meningiomas and particularly in the case of CPA, our experience and those of others showed that, when the treatment is discontinued, most of the tumors stabilize in size or decrease, avoiding the need for further surgery [7,8,11,12,15,16,20]. Thus, most authors now recommend careful follow-up instead of active treatment if urgent surgery is not needed.…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Atypical evolution of meningiomatosis after discontinuation of cyproterone acetate: clinical cases and histomolecular characterization

et al. 2021

View full text Add to dashboard Cite

Long-term use of cyproterone acetate (CPA) is associated with an increased risk of developing an intracranial meningioma. Discontinuation of CPA most often induce stabilization or regression of the tumor. The exact mechanism of regression is unknown as well as the reason why some meningiomas are still growing after CPA discontinuation.We are reporting four patients with multiple meningiomas, showing opposite tumor evolutions after stopping the CPA highlighting the underlying histologic and genetic features. MethodsPatients presenting several meningiomaswith opposite evolutions following the discontinuation of CPA were identi ed. The clinical and radiological data's were reviewed. A retrospective volumetric analysis of the meningiomas was performed. All the growing meningiomas were operated. Each tumor was characterized histopathologically and by molecular and genetic analyses. ResultsFour female withmultiple meningiomas and opposite tumor volume evolution after CPA discontinuation were identi ed. The histopathological results found broblastic meningiomas for tumorsgrowinglocated in the convexity and a more brousmeningioma in the skull-base tumor which decreased. Meningothelial and transitional meningiomaswere found in two skull-base growing meningiomas.The molecular characterization found twoNF2-mutations among the 4 growing meningiomas. In one patient who presented both patterns, the shrinking skull-basetumor harbored a PIK3CA-mutation whereas the growing tumor, a NF2-mutation. ConclusionTo our knowledge, this is the rst report of such an atypical tumor evolution of CPA-associated meningiomatosis after CPA discontinuation in the same patient. Underlying biological mechanisms explaining this observationespecially, the close relationship between mutational landscapes and the meningeal embryologyin CPA-related meningiomasrequire further research.

show abstract

“…This feature and a higher rate of multiple meningiomas suggest a strong relationship between this hormonal treatment and meningioma development. Therefore, delayed treatment of hormonal induced meningiomas with close clinical and MRI follow-up is recommended by several authors [3,8,9,13,19,22,23,26,28] in asymptomatic patients. Nevertheless, this management still needs further evaluation.…”

Section: Introductionmentioning

confidence: 99%

Have Progestin Related and Sporadic High Grade Meningiomas the Same Natural History?

Devalckeneer¹,

Aboukaïs²,

Faisant³

et al. 2021

Preprint

View full text Add to dashboard Cite

Introduction: High grade progestin related meningiomas have been reported in recent series but we found no previous study describing their long-term outcome. Our study aimed to evaluate patients operated on for high grade intracranial meningioma and who underwent long term exposure to high dose of cyproterone acetate, nomegestrol acetate and chlormadinone acetate.Methods: Our study retrospectively included 9 patients with high grade progestin related intracranial meningioma between December 2006 and December 2020. In each patient, clinico-radiological follow-up was performed every 6 months after diagnosis and treatment withdrawal recommendation. Results: The mean progestative exposure was 11.4 years. Edema existence or absence of cleft sign on MRI were the key factors for surgical indication. All patients underwent surgery. Ajduvant radiotherapy was indicated in 1 patient, and Gamma Knife Radiosurgery was proposed in 2 other patients for a second location of mengioma. 6 patients harbored a grade II chordoïd meningioma subtype with 100% PR expression and 3 patients a grade II atypical meningioma subtype with lower PR expression. The mean follow-up was 7.1 years and none of the 9 patients presented with a recurrence.Conclusion: Patients with Grade II progestin related meningiomas have less tumor recurrence after surgery than patients with sporadic high grade meningomas, especially after progestin withdrawal. The presence/ appearance of peri-meningioma edema and the absence of cleft sign before volumetric change should suggest the existence of an underlying high grade meningioma. In these cases, surgical resection may immediately be considered and adjuvant radiotherapy should be reserved for proven recurrence cases.

show abstract

Regression of giant olfactory groove meningioma and complete visual acuity recovery after discontinuation of cyproterone acetate

Cited by 21 publications

References 8 publications

WITHDRAWN: Nomegestrol Acetate or Chlormadinone Acetate Progestative Treatment in Women: Meningioma Behavior at Treatment Discontinuation

WITHDRAWN: Nomegestrol Acetate or Chlormadinone Acetate Progestative Treatment in Women: Meningioma Behavior at Treatment Discontinuation

Atypical evolution of meningiomatosis after discontinuation of cyproterone acetate: clinical cases and histomolecular characterization

Have Progestin Related and Sporadic High Grade Meningiomas the Same Natural History?

Contact Info

Product

Resources

About