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Postoperative speech impairment and cranial nerve deficits after secondary surgery of posterior fossa tumours in childhood: a prospective European multicentre study

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Abstract

Purpose

Brain tumours constitute 25% of childhood neoplasms, and half of them are in the posterior fossa. Surgery is a fundamental component of therapy, because gross total resection is associated with a higher progression-free survival. Patients with residual tumour, progression of residual tumour or disease recurrence commonly require secondary surgery. We prospectively investigated the risk of postoperative speech impairment (POSI) and cranial nerve dysfunction (CND) following primary and secondary resection for posterior cranial fossa tumours.

Methods

In the Nordic-European study of the cerebellar mutism syndrome, we prospectively included children undergoing posterior fossa tumour resection or open biopsy in one of the 26 participating European centres. Neurological status was assessed preoperatively, and surgical details were noted post-operatively. Patients were followed up 2 weeks, 2 months and 1 year postoperatively. Here, we analyse the risk of postoperative speech impairment (POSI), defined as either mutism or reduced speech, and cranial nerve dysfunction (CND) following secondary, as compared to primary, surgery.

Results

We analysed 426 children undergoing primary and 78 undergoing secondary surgery between 2014 and 2020. The incidence of POSI was significantly lower after secondary (12%) compared with primary (28%, p = 0.0084) surgery. In a multivariate analysis adjusting for tumour histology, the odds ratio for developing POSI after secondary surgery was 0.23, compared with primary surgery (95% confidence interval: 0.08–0.65, p = 0.006). The frequency of postoperative CND did not differ significantly after primary vs. secondary surgery (p = 0.21).

Conclusion

Children have a lower risk of POSI after secondary than after primary surgery for posterior fossa tumours but remain at significant risk of both POSI and CND. The present findings should be taken in account when weighing risks and benefits of secondary surgery for posterior fossa tumours.

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Acknowledgements

We thank the entire CMS-study group who took on the inclusion and follow-up of patients.

Funding

This study was funded by The Danish Childhood Cancer Foundation, The Swedish Childhood Cancer Foundation, The Brain Tumour Charity (UK), The Danish Cancer Society, King Christian IX and Queen Louise’s anniversary grant, The Danish Capitol Regions Research Fund, Dagmar Marshall foundation and Rigshospitalet's Research Fund in support of oncology purposes and Brainstrust (SCOT). Børnecancerfonden, 2012–9, 2014–3, 2015–14, 2016–0143, 2018–3653, 2020–6732, Barncancerfonden, PR2012-0106,KP2019-0014, Brain Tumour Charity, Kræftens Bekæmpelse, R124-A7920-15-S2, King Christian IX and Queen Louise’s anniversary grant, the Danish Capitol Regions Research Fund, Dagmar Marshall Foundation,Rigshospitalet’s Research Fund in support of oncology purposes, Braintrust. All research at Great Ormond Street Hospital NHS Foundation Trust and UCL Great Ormond Street Institute of Child Health is made possible by the NIHR Great Ormond Street Hospital Biomedical Research Centre. This work is part of Childhood Oncology Network Targeting Research, Organization & Life expectancy (CONTROL) and supported by Danish Cancer Society (R-257-A14720) and the Danish Childhood Cancer Foundation (2019–5934). The funding bodies had no role in the design of the study, analyses, interpretation of the data or decision to submit results.

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Authors and Affiliations

  1. Department of Neurosurgery, Copenhagen University Hospital Rigshospitalet, Blegdamsvej 9, 2100, Copenhagen E, Denmark

    J. Kjær Grønbæk & M. Juhler

  2. Department of Paediatrics and Adolescent Medicine, Copenhagen University Hospital Rigshospitalet, Blegdamsvej 9, 2100, Copenhagen E, Denmark

    J. Kjær Grønbæk, M. Wibroe, K. Nysom, K. Schmiegelow, A. Sehested & R. Mathiasen

  3. Department of Neurosurgery, Great Ormond Street Hospital for Children, Great Ormond Street, London, WC1N 3JH, UK

    S. Toescu & K. Aquilina

  4. Developmental Imaging and Biophysics Section, UCL-GOS Institute of Child Health, 30 Guilford St, London, WC1N 1EH, UK

    S. Toescu

  5. Department of Neurosurgery, Oslo University Hospital, Postboks 4950 Nydalen, 0424, Oslo, Norway

    R. Frič

  6. Department of Neuroscience, Section of Neurosurgery, Uppsala University Hospital, 751 85, Uppsala, Sweden

    P. Nilsson

  7. Department of Health Sciences, Lund University, Box 117, 221 00, Lund, Sweden

    C. Castor

  8. Department of Paediatric Neurosurgery, Alder Hey Children’s NHS Foundation Trust, E. Prescot Road, Liverpool, L14 5AB, UK

    C. Mallucci

  9. School of Health and Life Sciences, University of Liverpool, Brownlow Hill, Liverpool, L69 3BX, UK

    B. Pizer

  10. Department of Neurology, The Queen Elizabeth University Hospital, University of Glasgow, University Avenue, Glasgow, G12 8QQ, UK

    E. Molinari

  11. Department of Pediatric Hematology and Oncology, St Olavs Hospital, Postboks 3250 Torgarden, 7006, Trondheim, Norway

    M. Aasved Hjort

  12. Department of Neurosurgery, Helsinki University Hospital and University of Helsinki, P.O. Box 266, N00029, Helsinki, Finland

    A. Karppinen

  13. Department of Pediatrics, Lithuanian University of Health Science, Mickeviciaus 9, 44307, Kaunas, Lithuania

    G. Rutkauskiene

  14. 2Nd Department of Pediatrics, Semmelweis University, Tűzoltó street 7–9, Budapest, Hungary

    K. Mudra

  15. Neurology and Neurosurgery, National Institute of Mental Health, Laky Adolf u. 44, 1145, Budapest, Hungary

    B. Markia

  16. Princess Máxima Center for Pediatric Oncology, Heidelberglaan 25, 3584 CS, Utrecht, The Netherlands

    K. van Baarsen & E. Hoving

  17. Department of Neurosurgery, Pediatric Neurosurgery, University Hospital Tuebingen, Hoppe-Seyler-Str. 3, 72076, Tuebingen, Germany

    J. Zipfel

  18. Institute of Clinical Medicine, Faculty of Medicine, University of Copenhagen, Blegdamsvej 3B, 2200, Copenhagen, Denmark

    K. Schmiegelow & M. Juhler

  19. Department of Neurosurgery, Aarhus University Hospital, Palle Juul-Jensens, Boulevard 99, 8200, Aarhus, Denmark

    M. Juhler

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  1. J. Kjær Grønbæk
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The CMS study group

Contributions

All authors took part in the collection of data. JKG, AS, KS, MW, RM and MJ contributed to data analysis and interpretation. JKG, ST, RF, KS, AS, RM and MJ prepared the first draft of the manuscript. All authors approved the final version of the manuscript.

Corresponding author

Correspondence to J. Kjær Grønbæk.

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Ethics approval

This study was approved in Denmark by the Research Ethics Committees of the Capital Region (H-6–2014-002). The study was approved locally in all participating countries.

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The authors declare no financial, personal, or other conflict of interest.

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Grønbæk, J.K., Toescu, S., Frič, R. et al. 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, 747–758 (2022). https://doi.org/10.1007/s00381-022-05464-0

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