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Cerebellar mutism after posterior fossa tumor resection in children: a multicenter international retrospective study to determine possible modifiable factors

  • Bryan RenneEmail author
  • Julia Radic
  • Deepak Agrawal
  • Brittany Albrecht
  • Christopher M. Bonfield
  • Gesa Cohrs
  • Taylor Davis
  • Ashok Gupta
  • Andrea L. O. Hebb
  • Maria Lamberti-Pasculli
  • Friederike Knerlich-Lukoschus
  • Spencer Lindsay
  • P. Daniel McNeely
  • Shibu Pillai
  • Hitesh Inder Singh Rai
  • Katherine D. Sborov
  • Aleksander Vitali
  • Simon Walling
  • Peter Woerdeman
  • Wihasto Suryaningtyas
  • Douglas Cochrane
  • Ash Singhal
  • Paul Steinbok
Original Article

Abstract

Purpose

A preliminary survey of pediatric neurosurgeons working at different centers around the world suggested differences in clinical practice resulting in variation in the risk of pediatric cerebellar mutism (CM) and cerebellar mutism syndrome (CMS) after posterior fossa (PF) tumor resection. The purposes of this study were (1) to determine the incidence and severity of CM and CMS after midline PF tumor resection in children treated at these centers and (2) to identify potentially modifiable factors related to surgical management (rather than tumor biology) that correlate with the incidence of CM/CMS.

Methods

Attending pediatric neurosurgeons at British Columbia’s Children’s Hospital (BCCH) and neurosurgeons who completed a pediatric neurosurgery fellowship at BCCH were invited to provide data from the center where they currently practiced. Children aged from birth to less than 18 years who underwent initial midline PF tumor resection within a contemporary, center-selected 2-year period were included. Data was obtained by retrospective chart and imaging review. Modifiable surgical factors that were assessed included pre-resection surgical hydrocephalus treatment, surgical positioning, ultrasonic aspirator use, intraoperative external ventricular drain (EVD) use, surgical access route to the tumor, and extent of resection. CM was defined as decreased or absent speech output postoperatively and CMS as CM plus new or worsened irritability.

Results

There were 263 patients from 11 centers in 6 countries (Canada, Germany, the Netherlands, India, Indonesia, and the USA). Median age at surgery was 6 years (range < 1 to 17 years). The overall incidence of postoperative CM was 23.5% (range 14.7–47.6% for centers with data on ≥ 20 patients). The overall incidence of CMS was 6.5% (range 0–10.3% for centers contributing data on ≥ 20 patients). A multivariate logistic regression on the full data set showed no significant association between pre-resection surgical hydrocephalus treatment, prone position, ultrasonic aspirator use, EVD use, telovelar approach, complete or near total resection, or treating center and either postoperative CM or CMS.

Conclusions

While there was variation in surgical management of midline PF tumors among centers participating in this study, the factors in management that were examined did not predict postoperative CM or CMS.

Keywords

Cerebellar mutism syndrome Posterior fossa tumor Surgical management Hydrocephalus treatment Ultrasonic aspirator 

Notes

Compliance with ethical standards

Ethics approval for the study was granted by the University of British Columbia Children’s and Women’s Research Ethics Board (approvals CW16-067 and H15-03332).

Conflict of interest

None.

References

  1. 1.
    Avula S, Mallucci C, Kumar R, Pizer B (2015) Posterior fossa syndrome following brain tumour resection: review of pathophysiology and a new hypothesis on its pathogenesis. Childs Nerv Syst 31:1859–1867.  https://doi.org/10.1007/s00381-015-2797-0 CrossRefGoogle Scholar
  2. 2.
    Catsman-Berrevoets CE, Van Dongen HR, Mulder PG, Paz y Geuze D, Paquier PF, Lequin MH (1999) Tumour type and size are high risk factors for the syndrome of “cerebellar” mutism and subsequent dysarthria. J Neurol Neurosurg Psychiatry 67:755–757CrossRefGoogle Scholar
  3. 3.
    Doxey D, Bruce D, Sklar F, Swift D, Shapiro K (1999) Posterior fossa syndrome: identifiable risk factors and irreversible complications. Pediatr Neurosurg 31:131–136CrossRefGoogle Scholar
  4. 4.
    Gajjar A, Sanford RA, Bhargava R, Heideman R, Walter A, Li Y, Langston JW, Jenkins JJ, Muhlbauer M, Boyett J, Kun LE (1996) Medulloblastoma with brain stem involvement: the impact of gross total resection on outcome. Pediatr Neurosurg 25:182–187CrossRefGoogle Scholar
  5. 5.
    Griwan MS, Sharma BS, Mahajan RK, Kak VK (1993) Value of precraniotomy shunts in children with posterior fossa tumours. Childs Nerv Syst 9:462–465.  https://doi.org/10.1007/bf00393549 CrossRefGoogle Scholar
  6. 6.
    Gudrunardottir T, Morgan AT, Lux AL, Walker DA, Walsh KS, Wells EM, Wisoff JH, Juhler M, Schmahmann JD, Keating RF, Catsman-Berrevoets C (2016) Consensus paper on post-operative pediatric cerebellar mutism syndrome: the Iceland Delphi results. Childs Nerv Syst 32:1195–1203.  https://doi.org/10.1007/s00381-016-3093-3 CrossRefGoogle Scholar
  7. 7.
    Gudrunardottir T, Sehested A, Juhler M, Schmiegelow K (2011) Cerebellar mutism: incidence, risk factors and prognosis. Childs Nerv Syst 27:513–514; author reply 515.  https://doi.org/10.1007/s00381-010-1383-8 CrossRefGoogle Scholar
  8. 8.
    Kellogg JX, Piatt JH Jr (1997) Resection of fourth ventricle tumors without splitting the vermis: the cerebellomedullary fissure approach. Pediatr Neurosurg 27:28–33CrossRefGoogle Scholar
  9. 9.
    Korah MP, Esiashvili N, Mazewski CM, Hudgins RJ, Tighiouart M, Janss AJ, Schwaibold FP, Crocker IR, Curran WJ Jr, Marcus RB Jr (2010) Incidence, risks, and sequelae of posterior fossa syndrome in pediatric medulloblastoma. Int J Radiat Oncol Biol Phys 77:106–112.  https://doi.org/10.1016/j.ijrobp.2009.04.058 CrossRefGoogle Scholar
  10. 10.
    Law N, Greenberg M, Bouffet E, Taylor MD, Laughlin S, Strother D, Fryer C, McConnell D, Hukin J, Kaise C, Wang F, Mabbott DJ (2012) Clinical and neuroanatomical predictors of cerebellar mutism syndrome. Neuro-Oncology 14:1294–1303.  https://doi.org/10.1093/neuonc/nos160 CrossRefGoogle Scholar
  11. 11.
    Louis DN, Ohgaki H, Wiestler OD, Cavenee WK, Burger PC, Jouvet A, Scheithauer BW, Kleihues P (2007) The 2007 WHO classification of tumours of the central nervous system. Acta Neuropathol 114:97–109.  https://doi.org/10.1007/s00401-007-0243-4 CrossRefGoogle Scholar
  12. 12.
    Ostling L, Raffel C (2015) Editorial: complications following preresection shunting in patients with posterior fossa tumors. J Neurosurg Pediatr 15:1–3.  https://doi.org/10.3171/2014.9.Peds14440 CrossRefGoogle Scholar
  13. 13.
    Patay Z (2015) Postoperative posterior fossa syndrome: unraveling the etiology and underlying pathophysiology by using magnetic resonance imaging. Childs Nerv Syst 31:1853–1858.  https://doi.org/10.1007/s00381-015-2796-1 CrossRefGoogle Scholar
  14. 14.
    Pitsika M, Tsitouras V (2013) Cerebellar mutism. J Neurosurg Pediatr 12:604–614CrossRefGoogle Scholar
  15. 15.
    Renne B, Radic J, Agrawal D, Albrecht B, Bonfield C, Cohrs G, Davis T, Gupta A, Hebb ALO, Knerlich-Lukoschus F, Lindsay S, McNeely D, Pillai S, Rai HIS, Sborov KD, Vitali A, Walling S, Cochrane D, Singhal A, Steinbok P (2017) Cerebellar mutism after posterior fossa tumor resection in children: a multi-center international retrospective study to determine possible modifiable factors. In Proceedings of 45th Annual Meeting of International Society for Pediatric Neurosurgery, Denver, USA, 8–12 October, 2017. Childs Nerv Syst 33:1805.  https://doi.org/10.1007/s00381-017-3557-0
  16. 16.
    Robertson PL, Muraszko KM, Holmes EJ, Sposto R, Packer RJ, Gajjar A, Dias MS, Allen JC, Children's Oncology G (2006) Incidence and severity of postoperative cerebellar mutism syndrome in children with medulloblastoma: a prospective study by the Children’s Oncology Group. J Neurosurg 105:444–451.  https://doi.org/10.3171/ped.2006.105.6.444 Google Scholar
  17. 17.
    Sari E, Sari S, Akgun V, Ozcan E, Ince S, Babacan O, Saldir M, Acikel C, Basbozkurt G, Yesilkaya S, Kilic C, Kara K, Vurucu S, Kocaoglu M, Yesilkaya E (2015) Measures of ventricles and Evans index: from neonate to adolescent. Pediatr Neurosurg 50:12–17.  https://doi.org/10.1159/000370033 CrossRefGoogle Scholar
  18. 18.
    Steinbok P, Cochrane DD, Perrin R, Price A (2003) Mutism after posterior fossa tumour resection in children: incomplete recovery on long-term follow-up. Pediatr Neurosurg 39:179–183 72468CrossRefGoogle Scholar
  19. 19.
    Tamburrini G, Frassanito P, Chieffo D, Massimi L, Caldarelli M, Di Rocco C (2015) Cerebellar mutism. Childs Nerv Syst 31:1841–1851.  https://doi.org/10.1007/s00381-015-2803-6 CrossRefGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2019

Authors and Affiliations

  • Bryan Renne
    • 1
    Email author
  • Julia Radic
    • 2
  • Deepak Agrawal
    • 3
  • Brittany Albrecht
    • 4
  • Christopher M. Bonfield
    • 5
  • Gesa Cohrs
    • 6
  • Taylor Davis
    • 4
  • Ashok Gupta
    • 7
  • Andrea L. O. Hebb
    • 8
  • Maria Lamberti-Pasculli
    • 9
  • Friederike Knerlich-Lukoschus
    • 6
  • Spencer Lindsay
    • 4
  • P. Daniel McNeely
    • 8
  • Shibu Pillai
    • 10
  • Hitesh Inder Singh Rai
    • 3
  • Katherine D. Sborov
    • 5
  • Aleksander Vitali
    • 2
  • Simon Walling
    • 8
  • Peter Woerdeman
    • 11
  • Wihasto Suryaningtyas
    • 12
  • Douglas Cochrane
    • 9
  • Ash Singhal
    • 13
  • Paul Steinbok
    • 13
  1. 1.Faculty of MedicineUniversity of British ColumbiaVancouverCanada
  2. 2.Division of Neurosurgery, Royal University HospitalUniversity of SaskatchewanSaskatoonCanada
  3. 3.Department of Neurosurgery and Gamma KnifeAll India Institute of Medical SciencesNew DelhiIndia
  4. 4.University of Saskatchewan College of MedicineSaskatoonCanada
  5. 5.Department of NeurosurgeryVanderbilt University Medical CenterNashvilleUSA
  6. 6.Department of NeurosurgeryUniversity Hospital of Schleswig-Holstein, Campus KielKielGermany
  7. 7.Department of NeurosurgerySMS Medical CollegeJaipurIndia
  8. 8.Division of NeurosurgeryIWK Health CentreHalifaxCanada
  9. 9.Division of NeurosurgeryThe Hospital for Sick ChildrenTorontoCanada
  10. 10.Department of Neurosurgery, Mazumdar Shaw Medical CenterNarayana HealthBangaloreIndia
  11. 11.Department of Neurosurgery, University Medical Center UtrechtUtrecht UniversityUtrechtThe Netherlands
  12. 12.Department of Neurosurgery, Faculty of MedicineUniversitas Airlangga and Dr. Soetomo General HospitalSurabayaIndonesia
  13. 13.Division of Neurosurgery, Department of SurgeryBritish Columbia Children’s Hospital and University of British ColumbiaVancouverCanada

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