Tumour volume as a predictor of postoperative speech impairment in children undergoing resection of posterior fossa tumours: a prospective, multicentre study
Language English Country Austria Media electronic
Document type Journal Article, Multicenter Study
PubMed
40178678
PubMed Central
PMC11968465
DOI
10.1007/s00701-025-06459-x
PII: 10.1007/s00701-025-06459-x
Knihovny.cz E-resources
- Keywords
- CMS, PFS, Paediatrics, Posterior fossa tumour, Postoperative speech impairment, Volumetry,
- MeSH
- Child MeSH
- Infratentorial Neoplasms * surgery pathology MeSH
- Humans MeSH
- Magnetic Resonance Imaging MeSH
- Medulloblastoma * surgery pathology MeSH
- Adolescent MeSH
- Mutism * etiology MeSH
- Cerebellar Neoplasms * surgery pathology MeSH
- Postoperative Complications * etiology MeSH
- Speech Disorders * etiology MeSH
- Child, Preschool MeSH
- Prospective Studies MeSH
- Tumor Burden * MeSH
- Check Tag
- Child MeSH
- Humans MeSH
- Adolescent MeSH
- Male MeSH
- Child, Preschool MeSH
- Female MeSH
- Publication type
- Journal Article MeSH
- Multicenter Study MeSH
BACKGROUND: Cerebellar Mutism Syndrome (CMS) is a neurological complication of posterior fossa (PF) tumour surgery in children, and postoperative speech impairment (POSI) is the cardinal symptom of CMS. The role of tumour volume on the risk of POSI remains unexplored. This study investigates the association between tumour volume and the risk of POSI. METHODS: We included 360 patients from the European CMS study with available preoperative T1-weighted contrast-enhanced brain MRI. Speech status was assessed within two weeks postoperatively and categorised into three levels: habitual speech, severely reduced speech, and mutism. Tumour volumes were calculated using the BrainLab Elements SmartBrush™, a semi-automated segmentation tool. We used proportional odds models to estimate the odds ratio (OR) with adjustments for tumour location, pathology, and age. Based on the primary analysis, a risk stratification model for medulloblastoma patients was constructed, and the optimal volume cut-off was determined with Youden's Index. RESULTS: We found no effect of the overall tumour volume on the risk of POSI. This result did not change when adjusted for tumour location, pathology, and age. We found an association between tumour volume of medulloblastoma and the risk of POSI (unadjusted OR of 1.04 per increase in cm3 (95% CI 1.01;1.07, p = 0.01)), which did not change when adjusting for tumour location and age. The risk stratification cut-off for the tumour volume of medulloblastoma was calculated to be 16,5 cm3. Patients with medulloblastoma and preoperative tumour volumes below 16,5 cm3 had an absolute risk of 13% for POSI (low-risk group), whereas patients with preoperative tumour volumes above 16,5 cm3 had an absolute risk of 50% for POSI (high-risk group). CONCLUSION: Our data showed an association between preoperative tumour volume and the risk of POSI in children with medulloblastoma, while no association was found for the volume of other tumour types. We suggest a straightforward cut-off risk model for assessing the risk of POSI in children with medulloblastoma based on preoperative tumour volume. This approach can aid clinicians in informing patients and parents about the complications related to CMS following PF tumour surgery in children. CLINICAL TRIALS: ID NCT02300766 (October 2014).
2nd Department of Pediatrics Semmelweis University Budapest Hungary
Department of Neurosurgery 2nd Medical Faculty and Motol University Hospital Prague Czechia
Department of Neurosurgery Rigshospitalet Copenhagen Denmark
Department of Neurosurgery University Hospital Leipzig Leipzig Germany
Department of Neurosurgery University Hospital Oslo Oslo Norway
Department of Paediatrics and Adolescent Medicine Rigshospitalet Copenhagen Denmark
Department of Pediatric Hematology and Oncology St Olavs Hospital Trondheim Norway
Department of Pediatrics Lithuanian University of Health Science Kaunas Lithuania
Department of Radiology Alder Hey Children'S NHS Foundation Liverpool UK
National Institute of Neuroscience Budapest Hungary
Princess Maxima Center for Pediatric Oncology Utrecht Netherlands
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Avula S et al (2015) Posterior fossa syndrome following brain tumour resection: review of pathophysiology and a new hypothesis on its pathogenesis. Childs Nerv Syst. 31(10):1859–67. 10.1007/s00381-015-2797-0. PubMed
Avula S et al (2016) Post-operative pediatric cerebellar mutism syndrome and its association with hypertrophic olivary degeneration. Quant Imaging Med Surg. 6(5):535–544. 10.21037/qims.2016.10.11. PubMed PMC
Bae D, Mlc VV, Catsman-Berrevoets CE (2020) Preoperative prediction of postoperative cerebellar mutism syndrome. Validation of existing MRI models and proposal of the new Rotterdam pCMS prediction model. Childs Nerv Syst. 36(7):1471–1480. 10.1007/s00381-020-04535-4. PubMed PMC
Catsman-Berrevoets CE et al (1999) Tumour type and size are high risk factors for the syndrome of "cerebellar" mutism and subsequent dysarthria. J Neurol Neurosurg Psychiatry. 67(6):755–7. 10.1136/jnnp.67.6.755. PubMed PMC
Dixon L et al (2022) Diagnostic accuracy of qualitative MRI in 550 paediatric brain tumours: evaluating current practice in the computational era. Quant Imaging Med Surg. 12(1):131–143. 10.21037/qims-20-1388. PubMed PMC
Gronbaek JK et al (2021) Postoperative speech impairment and surgical approach to posterior fossa tumours in children: a prospective European multicentre cohort study. Lancet Child Adolesc Health. 5(11):814–824. 10.1016/S2352-4642(21)00274-1. PubMed
Gronbaek JK et al (2022) Left-handedness should not be overrated as a risk factor for postoperative speech impairment in children after posterior fossa tumour surgery: a prospective European multicentre study. Childs Nerv Syst. 38(8):1479–1485. 10.1007/s00381-022-05567-8. PubMed
Gronbaek JK et al (2022) Postoperative speech impairment and cranial nerve deficits after secondary surgery of posterior fossa tumours in childhood: a prospective European multicentre study. Childs Nerv Syst. 38(4): 747–758. 10.1007/s00381-022-05464-0. PubMed
Gudrunardottir T et al (2016) Consensus paper on post-operative pediatric cerebellar mutism syndrome: the Iceland Delphi results. Childs Nerv Syst. 32(7):1195–203. 10.1007/s00381-016-3093-3. PubMed
Huber T et al (2017) Reliability of Semi-Automated Segmentations in Glioblastoma. Clin Neuroradiol. 27(2):153–161. 10.1007/s00062-015-0471-2. PubMed
Katki HA (2019) Quantifying risk stratification provided by diagnostic tests and risk predictions: Comparison to AUC and decision curve analysis. Stat Med. 38(16):2943–2955. 10.1002/sim.8163. PubMed PMC
Khan RB et al (2021) Clinical features, neurologic recovery, and risk factors of postoperative posterior fossa syndrome and delayed recovery: a prospective study. Neuro Oncol. 23(9):1586–1596. 10.1093/neuonc/noab030. PubMed PMC
Kobets AJ et al (2024) Volumetric segmentation in the context of posterior fossa-related pathologies: a systematic review. Neurosurg Rev. 47(1):170. 10.1007/s10143-024-02366-4. PubMed PMC
Kothari RU et al (1996) The ABCs of measuring intracerebral hemorrhage volumes. Stroke. 27(8):1304–5. 10.1161/01.str.27.8.1304. PubMed
Kwak R, Kadoya S, Suzuki T (1983) Factors affecting the prognosis in thalamic hemorrhage. Stroke. 14(4):493–500. 10.1161/01.str.14.4.493. PubMed
Law N et al (2012) Clinical and neuroanatomical predictors of cerebellar mutism syndrome. Neuro Oncol. 14(10):1294–303. 10.1093/neuonc/nos160. PubMed PMC
Miller NG et al (2010) Cerebellocerebral diaschisis is the likely mechanism of postsurgical posterior fossa syndrome in pediatric patients with midline cerebellar tumors. AJNR Am J Neuroradiol. 31(2):288–94. 10.3174/ajnr.A1821. PubMed PMC
Morris EB et al (2009) Proximal dentatothalamocortical tract involvement in posterior fossa syndrome. Brain. 132(Pt 11):3087–95. 10.1093/brain/awp241. PubMed PMC
Ojemann JG et al (2013) Diffusion tensor imaging of the superior cerebellar peduncle identifies patients with posterior fossa syndrome. Childs Nerv Syst. 29(11):2071–7. 10.1007/s00381-013-2205-6. PubMed
Pettersson SD et al (2022) Risk factors for postoperative cerebellar mutism syndrome in pediatric patients: a systematic review and meta-analysis. J Neurosurg Pediatr. 29(4):467–475. 10.3171/2021.11.PEDS21445. PubMed
Pols S et al (2017) Risk factors for development of postoperative cerebellar mutism syndrome in children after medulloblastoma surgery. J Neurosurg Pediatr. 20(1):35–41. 10.3171/2017.2.PEDS16605. PubMed
Prasad KSV (2017) Clinicopathological Study of Pediatric Posterior Fossa Tumors. J Pediatr Neurosci 12(3):245-250. 10.4103/jpn.JPN_113_16 PubMed PMC
Renne B et al (2020) Cerebellar mutism after posterior fossa tumor resection in children: a multicenter international retrospective study to determine possible modifiable factors. Childs Nerv Syst. 36(6):1159–1169. 10.1007/s00381-019-04058-7. PubMed
Sagiuchi T et al (2001) Bilateral crossed cerebello-cerebral diaschisis and mutism after surgery for cerebellar medulloblastoma. Ann Nucl Med. 15(2):157–60. 10.1007/BF02988609. PubMed
Schreiber JE et al (2017) Posterior fossa syndrome and long-term neuropsychological outcomes among children treated for medulloblastoma on a multi-institutional, prospective study. Neuro Oncol. 19(12):1673–1682. 10.1093/neuonc/nox135. PubMed PMC
Sidpra J et al (2022) Improved prediction of postoperative pediatric cerebellar mutism syndrome using an artificial neural network. Neurooncol Adv. 4(1):vdac003. 10.1093/noajnl/vdac003. PubMed PMC
Toescu SM et al (2021) Fourth ventricle tumors in children: complications and influence of surgical approach. J Neurosurg Pediatr. 27(1):52–61. 10.3171/2020.6.PEDS2089. PubMed
Wibroe M et al (2017) Cerebellar mutism syndrome in children with brain tumours of the posterior fossa. BMC Cancer. 17(1):439. 10.1186/s12885-017-3416-0. PubMed PMC
Yecies D et al (2019) Posterior fossa syndrome and increased mean diffusivity in the olivary bodies. J Neurosurg Pediatr. 24(4):376–381. 10.3171/2019.5.PEDS1964. PubMed
ClinicalTrials.gov
NCT02300766