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Sporadic and neurofibromatosis type 2-associated meningioma in children and adolescents

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A Publisher Correction to this article was published on 29 August 2023

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Abstract

Purpose

Pediatric meningioma differs not only in its rare incidence from the adult meningioma, but also in its clinical characteristics. Many treatment approaches of pediatric meningioma are based on the study results of adult meningioma studies. The aim of this study was to explore the clinical and epidemiological characteristics of pediatric meningioma.

Methods

Data on pediatric patients diagnosed between 1982 and 2021 with NF2-associated or sporadic meningioma and recruited in the trials/registries HIT-ENDO, KRANIOPHARYNGEOM 2000/2007 and KRANIOPHARYNGEOM Registry 2019 were retrospectively analyzed for clinical characteristics, etiology, histology, therapy, and outcome.

Results

One hundred fifteen study participants were diagnosed with sporadic or NF2-associated meningioma at a median age of 10.6 years. There was a 1:1 sex ratio, with 14% of study participants suffering from NF2. 46% of the meningiomas were located hemispherically, 17% at the optic nerve/ intraorbital and 10% ventricularly. Multiple meningiomas were detected in 69% of NF2 patients and in 9% of sporadic meningiomas. 50% of the meningiomas were WHO grade I, 37% WHO grade II and 6% WHO grade III. Progressions or recurrences occurred after a median interval of 1.9 years. Eight patients (7%) died, 3 of them due to disease. The event-free survival was higher for WHO grade I than for WHO grade II meningioma patients (p = 0.008).

Conclusions

The major difference to the preceding literature could be found in the distribution of different WHO grades and their influence on event-free survival. Prospective studies are warranted to assess the impact of different therapeutic regimens.

Clinical trial registration numbers

NCT00258453; NCT01272622; NCT04158284.

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Data Availability

The datasets generated during the current study are available from the corresponding author on reasonable request.

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Acknowledgements

The authors would like to thank colleagues, patients, and their families for participating in our study.

Funding

This study was funded by a grant (H.L.M., DKS2014.13) of the German Childhood Cancer Foundation, Bonn, Germany.

Author information

Authors and Affiliations

  1. Department of Pediatrics and Pediatric Hematology/Oncology, University Children’s Hospital, Carl von Ossietzky University of Oldenburg, Klinikum Oldenburg AöR, 26133, Oldenburg, Germany

    Katharina Wagener, Julia Beckhaus, Svenja Boekhoff, Carsten Friedrich & Hermann L. Müller

  2. Division of Epidemiology and Biometry, Carl von Ossietzky University of Oldenburg, 26129, Oldenburg, Germany

    Julia Beckhaus

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  1. Katharina Wagener
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  2. Julia Beckhaus
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  3. Svenja Boekhoff
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  4. Carsten Friedrich
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  5. Hermann L. Müller
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Contributions

All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by Katharina Wagener, Svenja Boekhoff, Carsten Friedrich and Julia Beckhaus. The first draft of the manuscript was written by Katharina Wagener and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Hermann L. Müller.

Ethics declarations

Competing interests

H.L.M. has received reimbursement of participation fees for scientific meetings and continuing medical education events from the following companies: Ferring, Lilly, Pfizer, Sandoz/Hexal, Novo Nordisk, IPSEN, and Merck Serono. He has received reimbursement of travel expenses from IPSEN and lecture honoraria from Pfizer. The other authors declare that they have no conflict of interest.

Ethics approval

All procedures performed in our study were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. The studies KRANIOPHARYNGEOM 2000 (Clinical trial registration number: NCT00258453) and KRANIOPHARYNGEOM 2007 (Clinical trial registration number: NCT01272622) were approved by the local standing-committee on ethical practice of the Medizinische Fakultät, Julius-Maximilians-Universität Würzburg, Germany (140/99; 94/06, respectively). Approval of the KRANIOPHARYNGEOM Registry 2019 and our meningioma study was granted by the Ethics Committee of Medical Faculty of the Carl von Ossietzky University of Oldenburg, Oldenburg, Germany (No: 2019-046, Date. 12/06/2019; No: 2021 − 166, Date 16/12/2021).

Consent to participate

Written parental and/or patient consent was obtained from all individual participants and their parents included in the study.

Consent for publication

Written consent for publication of results based on analyses of anonymized data was obtained from individual participants and/or legal guardians.

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Wagener, K., Beckhaus, J., Boekhoff, S. et al. Sporadic and neurofibromatosis type 2-associated meningioma in children and adolescents. J Neurooncol 163, 555–563 (2023). https://doi.org/10.1007/s11060-023-04344-0

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