Abstract
The aim of this study was to explore the association between sex and cerebellar mutism syndrome and to examine other potential risk factors. This ambispective cohort study examined 218 pediatric patients (132 boys) with a posterior fossa tumor who underwent tumor resection from July 2013 to March 2021. The patients’ demographics and tumor characteristics were examined and statistically analyzed to explore the associations among the variables. Multivariable and subgroup analyses were conducted to validate the independent risk factors for cerebellar mutism syndrome (CMS). The male and female patients did not differ significantly in terms of age, tumor size, tumor location, tumor consistency, VP shunt placement before resection, extent of resection, or surgeon, as well as with respect to the presence of hydrocephalus or paraventricular edema. The overall incidence of CMS was 32.6%. The incidence of CMS was significantly higher in male patients than that in female patients (41.7% vs. 18.6%; P = 0.001). In the multivariable analysis, male sex (adjusted odds ratio [OR], 3.27; P = 0.001), solid tumor consistency (adjusted OR, 5.61; P = 0.001), midline location (adjusted OR, 3.78; P = 0.004), and hydrocephalus (adjusted OR, 2.56; P = 0.047) were independent risk factors for the CMS. Chi-square analysis revealed that solid tumor consistency and midline location were associated with medulloblastoma (P < 0.001). Male patients had a higher risk of developing CMS after a posterior fossa tumor resection. Midline location, solid tumor consistency, and hydrocephalus were independent risk factors for CMS.
Similar content being viewed by others
Data Availability
Data is available at request.
References
Gudrunardottir T, Morgan AT, Lux AL, Walker DA, Walsh KS, Wells EM, et al. Consensus paper on post-operative pediatric cerebellar mutism syndrome: the Iceland Delphi results. Childs Nerv Syst. 2016;32:1195–203.
Gudrunardottir T, Sehested A, Juhler M, Schmiegelow K. Cerebellar mutism: review of the literature. Childs Nerv Syst. 2011;27:355–63.
Khan RB, Patay Z, Klimo P, Huang J, Kumar R, Boop FA, et al. Clinical features, neurologic recovery, and risk factors of postoperative posterior fossa syndrome and delayed recovery: a prospective study. Neuro Oncol. 2021;23:1586–96.
Morris EB, Phillips NS, Laningham FH, Patay Z, Gajjar A, Wallace D, et al. Proximal dentatothalamocortical tract involvement in posterior fossa syndrome. Brain. 2009;132:3087–95.
Grønbæk J, Molinari E, Avula S, Wibroe M, Oettingen G, Juhler M. The supplementary motor area syndrome and the cerebellar mutism syndrome: a pathoanatomical relationship? Childs Nerv Syst. 2020;36:1197–204.
Palesi F, Tournier JD, Calamante F, Muhlert N, Castellazzi G, Chard D, et al. Contralateral cerebello-thalamo-cortical pathways with prominent involvement of associative areas in humans in vivo. Brain Struct Funct. 2015;220:3369–84.
Palesi F, De Rinaldis A, Castellazzi G, Calamante F, Muhlert N, Chard D, et al. Contralateral cortico-ponto-cerebellar pathways reconstruction in humans in vivo: implications for reciprocal cerebro-cerebellar structural connectivity in motor and non-motor areas. Sci Rep. 2017;7:12841.
Marien P, Manto M. Cerebellum as a master-piece for linguistic predictability. Cerebellum. 2018;17:101–3.
Guell X, Gabrieli JDE, Schmahmann JD. Triple representation of language, working memory, social and emotion processing in the cerebellum: convergent evidence from task and seed-based resting-state fmri analyses in a single large cohort. Neuroimage. 2018;172:437–49.
Robertson PL, Muraszko KM, Holmes EJ, Sposto R, Packer RJ, Gajjar A, et al. Incidence and severity of postoperative cerebellar mutism syndrome in children with medulloblastoma: a prospective study by the children’s oncology group. J Neurosurg. 2006;105:444.
Kupeli S, Yalcin B, Bilginer B, Akalan N, Haksal P, Buyukpamukcu M. Posterior fossa syndrome after posterior fossa surgery in children with brain tumors. Pediatr Blood Cancer. 2011;56:206–10.
Toescu SM, Hales PW, Aquilina K, Clark CA. Quantitative MRI in post-operative paediatric cerebellar mutism syndrome. Eur J Radiol. 2018;108:43–51.
Bakhshi SK, Mitha R, Mushtaq N, Shamim MS. Cerebellar mutism syndrome after surgical resection of posterior fossa neoplastic lesions. J Pak Med Assoc. 2020;70:1667–8.
Ashida R, Nazar N, Edwards R, Teo M. Cerebellar mutism syndrome: an overview of the pathophysiology in relation to the cerebrocerebellar anatomy, risk factors, potential treatments, and outcomes. World Neurosurg. 2021;153:63–74.
Renne B, Radic J, Agrawal D, Albrecht B, Bonfield CM, Cohrs G, et al. Cerebellar mutism after posterior fossa tumor resection in children: a multicenter international retrospective study to determine possible modifiable factors. Childs Nerv Syst. 2020;36:1159–69.
Wibroe M, Cappelen J, Castor C, Clausen N, Grillner P, Gudrunardottir T, et al. Cerebellar mutism syndrome in children with brain tumours of the posterior fossa. BMC Cancer. 2017;17:439.
Catsman-Berrevoets CE. Cerebellar mutism syndrome: cause and rehabilitation. Curr Opin Neurol. 2017;30:133–9.
Cámara S, Fournier MC, Cordero P, Melero J, Robles F, Esteso B, et al. Neuropsychological profile in children with posterior fossa tumors with or without postoperative cerebellar mutism syndrome (cms). Cerebellum. 2020;19:78–88.
Jabarkheel R, Amayiri N, Yecies D, Huang Y, Toescu S, Nobre L, et al. Molecular correlates of cerebellar mutism syndrome in medulloblastoma. Neuro Oncol. 2020;22:290–7.
Gora NK, Gupta A, Sinha VD. Cerebellar mutism syndrome following midline posterior fossa tumor resection in children: an institutional experience. J Pediatr Neurosci. 2017;12:313–9.
Tomasi D, Volkow ND. Laterality patterns of brain functional connectivity: gender effects. Cereb Cortex. 2012;22:1455–62.
Dinga S, Wu D, Huang S, Wu C, Wang X, Shi J, et al. Neuromagnetic correlates of audiovisual word processing in the developing brain. Int J Psychophysiol. 2018;128:7–21.
Yu VY, MacDonald MJ, Oh A, Hua GN, De Nil LF, Pang EW. Age-related sex differences in language lateralization: a magnetoencephalography study in children. Dev Psychol. 2014;50:2276–84.
Adani S, Cepanec M. Sex differences in early communication development: behavioral and neurobiological indicators of more vulnerable communication system development in boys. Croat Med J. 2019;60:141–9.
Narzisi A, Posada M, Barbieri F, Chericoni N, Ciuffolini D, Pinzino M, et al. Prevalence of autism spectrum disorder in a large Italian catchment area: a school-based population study within the ASDEU project. Epidemiol Psychiatr Sci. 2018;29:e5.
Kogan MD, Vladutiu CJ, Schieve LA, Ghandour RM, Blumberg SJ, Zablotsky B, et al. The prevalence of parent-reported autism spectrum disorder among us children. Pediatrics. 2018;142,6:e20174161.
Pettersson SD, Kitlinski M, Miekisiak G, Ali S, Krakowiak M, Szmuda T. Risk factors for postoperative cerebellar mutism syndrome in pediatric patients: a systematic review and meta-analysis. J Neurosurg Pediatr. 2022;29:467–75.
Lange BP, Euler HA, Zaretsky E. Sex differences in language competence of 3- to 6-year-old children. Appl Psycholinguist. 2016;37:1417–38.
Ozcaliskan S, Goldin-Meadow S. Sex differences in language first appear in gesture. Dev Sci. 2010;13:752–60.
Pettersson SD, Kitlinski M, Miękisiak G, Ali S, Krakowiak M, Szmuda T. Risk factors for postoperative cerebellar mutism syndrome in pediatric patients: a systematic review and meta-analysis. J Neurosurg: Pediatr. 2021;5:1–9.
Gronbaek JK, Wibroe M, Toescu S, Fric R, Thomsen BL, Moller LN, et al. Postoperative speech impairment and surgical approach to posterior fossa tumours in children: a prospective european multicentre cohort study. Lancet Child Adolesc Health. 2021;5:814–24.
Cao F, Brennan C, Booth JR. The brain adapts to orthography with experience: evidence from English and Chinese. Dev Sci. 2015;18:785–98.
Ip KI, Marks RA, Hsu LS, Desai N, Kuan JL, Tardif T, et al. Morphological processing in Chinese engages left temporal regions. Brain Lang. 2019;199:104696.
Lou Y, Zhao L, Yu S, Sun B, Hou Z, Zhang Z, et al. Brain asymmetry differences between Chinese and Caucasian populations: a surface-based morphometric comparison study. Brain Imaging Behav. 2020;14:2323–32.
Doxey D, Bruce D, Sklar F, Swift D, Shapiro K. Posterior fossa syndrome: identifiable risk factors and irreversible complications. Pediatr Neurosurg. 1999;31:131–6.
Law N, Greenberg M, Bouffet E, Taylor MD, Laughlin S, Strother D, et al. Clinical and neuroanatomical predictors of cerebellar mutism syndrome. Neuro Oncol. 2012;14:1294–303.
Pols S, van Veelen M, Aarsen FK, Gonzalez CA, Catsman-Berrevoets CE. Risk factors for development of postoperative cerebellar mutism syndrome in children after medulloblastoma surgery. J Neurosurg Pediatr. 2017;20:35–41.
Tamburrini G, Frassanito P, Chieffo D, Massimi L, Caldarelli M, Di Rocco C. Cerebellar mutism. Childs Nerv Syst. 2015;31:1841–51.
Toescu SM, Samarth G, Layard HH, Issitt R, Margetts B, Phipps KP, et al. Fourth ventricle tumors in children: complications and influence of surgical approach. J Neurosurg Pediatr. 2020;27:52–61.
Acknowledgements
We thank Professor Yaguang Peng, who guided us in the statistical analysis.
Funding
This study was funded by Beijing Hospital’s Authority Clinical Medicine Development of Special Funding (code: XMLX202144).
Author information
Authors and Affiliations
Contributions
Conceptualization: Wei Yang, Ming Ge
Methodology: Wei Yang, Kaiyi Zhu
Formal analysis and investigation: Jiashu Chen, Ping Yang, Yingjie Cai, Xiaojiao Peng, Jia Wang
Writing–original draft preparation: Wei Yang
Writing–review and editing: Wei Yang, Kaiyi Zhu
Funding acquisition: Ming Ge
Supervision: Ming Ge
Resources: Hailang Sun, Yuanqi Ji, Fengmao Zhao, Hong Zhang
Corresponding author
Ethics declarations
Conflict of Interest
The authors declare no competing interests.
Ethics Approval
This study was performed in line with the principles of the Declaration of Helsinki. Approval was granted by the Ethics Committee of Beijing Children’s Hospital (Beijing, China, grant number: [2021]-E-024-Y).
Consent to Participate
Informed consent was obtained from all individual participants included in the study.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Yang, W., Ge, M., Zhu, K. et al. Male Predisposition in Cerebellar Mutism Syndrome: a Cohort Study. Cerebellum 22, 730–738 (2023). https://doi.org/10.1007/s12311-022-01449-6
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12311-022-01449-6