Abstract
Objective
Pediatric meningiomas are relatively rare and have atypical clinical features compared to adults. The purpose of this work is to report our 15-year experience in the management of pediatric meningiomas and assess their clinical characteristics, pathological features, and prognostic factors.
Methods
A total of 40 children (age ≤ 15 years) who were diagnosed as pediatric meningiomas were enrolled in this study. Patient information including clinical presentation, gender, age at time of diagnosis, histopathological features, tumor location, tumor volume, treatment methods, and follow-up data were extracted and analyzed.
Results
The mean age at diagnosis was 10.78 ± 3.50 years (range 2–15 years) in 40 patients with a male to female ratio of 1:1.11. Headache, epilepsy, visual disturbance, and limb weakness are common clinical manifestations. Two patients had multiple intracranial meningiomas. Fourteen (33.3%) of pediatric meningiomas were high grade meningiomas. Seven patients (17.5%) were treated with STR, while GTR was achieved in 33 patients (82.5%). The mean follow-up period was 82.1 months (range 9–173 months). Recurrence occurred in 9 patients (22.5%), and 5 patients (12.5%) passed away.
Conclusion
The incidence of pediatric meningiomas increases with advancing age. In pediatric patients, the percentage of high-grade tumors is higher than adults. Younger children were more likely to have high-grade meningiomas, while patients with tumors located in skull base or parasagittal/falx tend to have low-grade meningiomas. The WHO grade III meningiomas were significantly correlated with poor prognosis. Adjuvant radiotherapy after surgery can improve prognosis and may be a potential treatment strategy in children with malignant meningiomas.
Similar content being viewed by others
References
Ostrom QT, Gittleman H, Truitt G, Boscia A, Kruchko C, Barnholtz-Sloan JS (2018) CBTRUS Statistical report: primary brain and other central nervous system tumors diagnosed in the United States in 2011–2015. Neuro Oncol 20: iv1-iv86. https://doi.org/10.1093/neuonc/noy131
Louis DN, Perry A, Reifenberger G, von Deimling A, Figarella-Branger D, Cavenee WK, Ohgaki H, Wiestler OD, Kleihues P, Ellison DW (2016) The 2016 World Health Organization Classification of Tumors of the Central Nervous System: a summary. Acta Neuropathol 131:803–820. https://doi.org/10.1007/s00401-016-1545-1
Gao X, Zhang R, Mao Y, Wang Y (2009) Childhood and juvenile meningiomas. Childs Nerv Syst 25:1571–1580. https://doi.org/10.1007/s00381-009-0964-x
Greene S, Nair N, Ojemann JG, Ellenbogen RG, Avellino AM (2008) Meningiomas in children. Pediatr Neurosurg 44:9–13. https://doi.org/10.1159/000110656
Tufan K, Dogulu F, Kurt G, Emmez H, Ceviker N, Baykaner MK (2005) Intracranial meningiomas of childhood and adolescence. Pediatr Neurosurg 41:1–7. https://doi.org/10.1159/000084858
Perry A, Dehner LP (2003) Meningeal tumors of childhood and infancy. An update and literature review. Brain Pathol 13:386–408. https://doi.org/10.1111/j.1750-3639.2003.tb00038.x
Miranda P, Simal JA, Vila M, Hernandez M, Menor F, Alvarez-Garijo JA (2009) Posterior fossa clear cell meningioma without dural attachment in a child. Childs Nerv Syst 25:389–392. https://doi.org/10.1007/s00381-008-0757-7
Gump WC (2015) Meningiomas of the pediatric skull base: a review. J Neurol Surg B Skull Base 76:66–73. https://doi.org/10.1055/s-0034-1390012
Stanuszek A, Piatek P, Kwiatkowski S, Adamek D (2014) Multiple faces of children and juvenile meningiomas: a report of single-center experience and review of literature. Clin Neurol Neurosurg 118:69–75. https://doi.org/10.1016/j.clineuro.2013.12.019
Teixidor P, Guillen A, Cruz O, Costa JM (2008) Intracranial meningiomas in children: report of 10 cases. Neurocirugia (Astur) 19:434–439
Li Z, Li H, Jiao Y, Ma J, Wang S, Zhao J, Cao Y (2017) A comparison of clinicopathological features and surgical outcomes between pediatric skull base and non-skull base meningiomas. Childs Nerv Syst 33:595–600. https://doi.org/10.1007/s00381-016-3278-9
Kotecha RS, Junckerstorff RC, Lee S, Cole CH, Gottardo NG (2011) Pediatric meningioma: current approaches and future direction. J Neurooncol 104:1–10. https://doi.org/10.1007/s11060-010-0503-3
Hatef J, Adamson C, Obiga O, Taremwa B, Ssenyojo H, Muhumuza M, Haglund M, Schroeder K (2014) Central nervous system tumor distribution at a tertiary referral center in Uganda. World Neurosurg 82:258–265. https://doi.org/10.1016/j.wneu.2014.06.040
Kotecha RS, Pascoe EM, Rushing EJ, Rorke-Adams LB, Zwerdling T, Gao X, Li X, Greene S, Amirjamshidi A, Kim SK, Lima MA, Hung PC, Lakhdar F, Mehta N, Liu Y, Devi BI, Sudhir BJ, Lund-Johansen M, Gjerris F, Cole CH, Gottardo NG (2011) Meningiomas in children and adolescents: a meta-analysis of individual patient data. Lancet Oncol 12:1229–1239. https://doi.org/10.1016/S1470-2045(11)70275-3
Dudley RWR, Torok MR, Randall S, Beland B, Handler MH, Mulcahy-Levy JM, Liu AK, Hankinson TC (2018) Pediatric versus adult meningioma: comparison of epidemiology, treatments, and outcomes using the Surveillance, Epidemiology, and End Results database. J Neurooncol 137:621–629. https://doi.org/10.1007/s11060-018-2756-1
Ravindranath K, Vasudevan MC, Pande A, Symss N (2013) Management of pediatric intracranial meningiomas: an analysis of 31 cases and review of literature. Childs Nerv Syst 29:573–582. https://doi.org/10.1007/s00381-012-2010-7
Thuijs NB, Uitdehaag BM, Van Ouwerkerk WJ, van der Valk P, Vandertop WP, Peerdeman SM (2012) Pediatric meningiomas in The Netherlands 1974–2010: a descriptive epidemiological case study. Childs Nerv Syst 28:1009–1015. https://doi.org/10.1007/s00381-012-1759-z
Grossbach AJ, Mahaney KB, Menezes AH (2017) Pediatric meningiomas: 65-year experience at a single institution. J Neurosurg Pediatr 20:42–50. https://doi.org/10.3171/2017.2.PEDS16497
Liu Y, Li F, Zhu S, Liu M, Wu C (2008) Clinical features and treatment of meningiomas in children: report of 12 cases and literature review. Pediatr Neurosurg 44:112–117. https://doi.org/10.1159/000113112
Molleston MC, Moran CJ, Roth KA, Rich KM (1994) Infantile meningioma. Pediatr Neurosurg 21:195–200. https://doi.org/10.1159/000120835
El Beltagy MA, Enayet AE, Atteya MME, Reda M, Refaat A, Taha H, Ahmed S, Abdelaziz A (2019) Management of pediatric CNS meningiomas: CCHE-57357 experience in 39 cases. Childs Nerv Syst 35:1323–1331. https://doi.org/10.1007/s00381-019-04156-6
Santos MV, Furlanetti L, Valera ET, Brassesco MS, Tone LG, de Oliveira RS (2012) Pediatric meningiomas: a single-center experience with 15 consecutive cases and review of the literature. Childs Nerv Syst 28:1887–1896. https://doi.org/10.1007/s00381-012-1823-8
Lakhdar F, Arkha Y, El Ouahabi A, Melhaoui A, Rifi L, Derraz S, El Khamlichi A (2010) Intracranial meningioma in children: different from adult forms? A series of 21 cases. Neurochirurgie 56:309–314. https://doi.org/10.1016/j.neuchi.2010.05.008
Mehta N, Bhagwati S, Parulekar G (2009) Meningiomas in children: A study of 18 cases. J Pediatr Neurosci 4:61–65. https://doi.org/10.4103/1817-1745.57322
Caroli E, Russillo M, Ferrante L (2006) Intracranial meningiomas in children: report of 27 new cases and critical analysis of 440 cases reported in the literature. J Child Neurol 21:31–36. https://doi.org/10.1177/08830738060210010801
Dash C, Pasricha R, Gurjar H, Singh PK, Sharma BS (2016) Pediatric intraventricular meningioma: a series of six cases. J Pediatr Neurosci 11:193–196. https://doi.org/10.4103/1817-1745.193356
Deen HG Jr, Scheithauer BW, Ebersold MJ (1982) Clinical and pathological study of meningiomas of the first two decades of life. J Neurosurg 56:317–322. https://doi.org/10.3171/jns.1982.56.3.0317
Menon G, Nair S, Sudhir J, Rao BR, Mathew A, Bahuleyan B (2009) Childhood and adolescent meningiomas: a report of 38 cases and review of literature. Acta Neurochir (Wien) 151: 239–244; discussion 244. https://doi.org/10.1007/s00701-009-0206-8
Odebode TO, Akang EE, Shokunbi MT, Malamo AO, Ogunseyinde AO (2006) Factors influencing visual and clinical outcome in Nigerian patients with cranial meningioma. J Clin Neurosci 13:649–654. https://doi.org/10.1016/j.jocn.2005.07.023
Merten DF, Gooding CA, Newton TH, Malamud N (1974) Meningiomas of childhood and adolescence. J Pediatr 84:696–700. https://doi.org/10.1016/s0022-3476(74)80011-9
Modha A, Gutin PH (2005) Diagnosis and treatment of atypical and anaplastic meningiomas: a review. Neurosurgery 57: 538–550; discussion 538–550. https://doi.org/10.1227/01.neu.0000170980.47582.a5
Zhou P, Ma W, Yin S, Li Y, Jiang S (2013) Three risk factors for WHO grade II and III meningiomas: a study of 1737 cases from a single center. Neurol India 61:40–44. https://doi.org/10.4103/0028-3886.107928
He W, Liu Z, Teng H, Tang L, Jie D, Duan Z, Yu H, Xu J (2020) Pediatric meningiomas: 10-year experience with 39 patients. J Neurooncol 149:543–553. https://doi.org/10.1007/s11060-020-03649-8
Hui M, Uppin MS, Saradhi MV, Sahu BP, Purohit AK, Sundaram C (2015) Pediatric meningiomas an aggressive subset: a clinicopathological and immunohistochemical study. J Postgrad Med 61:32–35. https://doi.org/10.4103/0022-3859.147035
Kane AJ, Sughrue ME, Rutkowski MJ, Shangari G, Fang S, McDermott MW, Berger MS, Parsa AT (2011) Anatomic location is a risk factor for atypical and malignant meningiomas. Cancer 117:1272–1278. https://doi.org/10.1002/cncr.25591
Kuster O, Schittenhelm J, Schwartz O, Meyermann R, Schuhmann MU (2012) Paediatric clear cell meningioma with multiple distant recurrences after presumed intra-operative cell spread. Childs Nerv Syst 28:925–931. https://doi.org/10.1007/s00381-011-1669-5
Larrew T, Eskandari R (2016) Pediatric Intraparenchymal Meningioma: Case Report and Comparative Review. Pediatr Neurosurg 51:83–86. https://doi.org/10.1159/000441008
Sade B, Chahlavi A, Krishnaney A, Nagel S, Choi E, Lee JH (2007) World Health Organization Grades II and III meningiomas are rare in the cranial base and spine. Neurosurgery 61: 1194–1198; discussion 1198. https://doi.org/10.1227/01.neu.0000306097.38141.65
Kepes JJ (1986) Presidential address: the histopathology of meningiomas. A reflection of origins and expected behavior? J Neuropathol Exp Neurol 45:95–107
Lee JH, Sade B, Choi E, Golubic M, Prayson R (2006) Meningothelioma as the predominant histological subtype of midline skull base and spinal meningioma. J Neurosurg 105:60–64. https://doi.org/10.3171/jns.2006.105.1.60
Li Z, Li H, Jiao Y, Ma J, Wang S, Cao Y, Zhao J (2018) Clinical features and long-term outcomes of pediatric intraventricular meningiomas: data from a single neurosurgical center. Neurosurg Rev 41:525–530. https://doi.org/10.1007/s10143-017-0884-2
Dziuk TW, Woo S, Butler EB, Thornby J, Grossman R, Dennis WS, Lu H, Carpenter LS, Chiu JK (1998) Malignant meningioma: an indication for initial aggressive surgery and adjuvant radiotherapy. J Neurooncol 37:177–188. https://doi.org/10.1023/a:1005853720926
Toland A, McNulty SN, Pekmezci M, Evenson M, Huntoon K, Pierson CR, Boue DR, Perry A, Dahiya S (2020) Pediatric meningioma: a clinicopathologic and molecular study with potential grading implications. Brain Pathol 30:1134–1143. https://doi.org/10.1111/bpa.12884
McNulty SN, Schwetye K, Goldstein M, Carter J, Schmidt RE, Ansstas G, Tsien CI, Kim AH, Dahiya S (2018) Analysis of point mutations and copy number variation in Grade II and III meningioma. Exp Mol Pathol 105:328–333. https://doi.org/10.1016/j.yexmp.2018.10.007
Katz LM, Hielscher T, Liechty B, Silverman J, Zagzag D, Sen R, Wu P, Golfinos JG, Reuss D, Neidert MC, Wirsching HG, Baumgarten P, Herold-Mende C, Wick W, Harter PN, Weller M, von Deimling A, Snuderl M, Sen C, Sahm F (2018) Loss of histone H3K27me3 identifies a subset of meningiomas with increased risk of recurrence. Acta Neuropathol 135:955–963. https://doi.org/10.1007/s00401-018-1844-9
Clark VE, Erson-Omay EZ, Serin A, Yin J, Cotney J, Ozduman K, Avsar T, Li J, Murray PB, Henegariu O, Yilmaz S, Gunel JM, Carrion-Grant G, Yilmaz B, Grady C, Tanrikulu B, Bakircioglu M, Kaymakcalan H, Caglayan AO, Sencar L, Ceyhun E, Atik AF, Bayri Y, Bai H, Kolb LE, Hebert RM, Omay SB, Mishra-Gorur K, Choi M, Overton JD, Holland EC, Mane S, State MW, Bilguvar K, Baehring JM, Gutin PH, Piepmeier JM, Vortmeyer A, Brennan CW, Pamir MN, Kilic T, Lifton RP, Noonan JP, Yasuno K, Gunel M (2013) Genomic analysis of non-NF2 meningiomas reveals mutations in TRAF7, KLF4, AKT1, and SMO. Science 339:1077–1080. https://doi.org/10.1126/science.1233009
Battu S, Kumar A, Pathak P, Purkait S, Dhawan L, Sharma MC, Suri A, Singh M, Sarkar C, Suri V (2018) Clinicopathological and molecular characteristics of pediatric meningiomas. Neuropathology 38:22–33. https://doi.org/10.1111/neup.12426
Funding
This work was supported by National Natural Science Foundation of China (grant no. 81702478 and 81602204).
Author information
Authors and Affiliations
Corresponding authors
Ethics declarations
Conflict of interest
The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Jingdian Liu and Kai Zhao contributed equally in this work.
Supplementary information
Below is the link to the electronic supplementary material.
Rights and permissions
About this article
Cite this article
Liu, J., Zhao, K., Wang, J. et al. Clinical features and long-term outcomes of pediatric meningiomas. Childs Nerv Syst 37, 3041–3047 (2021). https://doi.org/10.1007/s00381-021-05296-4
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00381-021-05296-4