A comparison study assessing neuropsychological outcome of patients with post-operative pediatric cerebellar mutism syndrome and matched controls after proton radiation therapy

Julie A Grieco; Annah N Abrams; Casey L Evans; Torunn I Yock; Margaret B Pulsifer

doi:10.1007/s00381-019-04299-6

A comparison study assessing neuropsychological outcome of patients with post-operative pediatric cerebellar mutism syndrome and matched controls after proton radiation therapy

Childs Nerv Syst. 2020 Feb;36(2):305-313. doi: 10.1007/s00381-019-04299-6. Epub 2019 Jul 19.

Authors

Julie A Grieco^{1

2}, Annah N Abrams^{2

3}, Casey L Evans¹, Torunn I Yock^{2

4}, Margaret B Pulsifer^{5

6}

Affiliations

¹ Department of Psychiatry, Massachusetts General Hospital, One Bowdoin Square, 7th Floor, Boston, MA, 02114, USA.
² Harvard Medical School, Boston, MA, USA.
³ Department of Psychiatry, Massachusetts General Hospital, 55 Fruit Street, Boston, MA, 02114, USA.
⁴ Department of Radiation Oncology, Massachusetts General Hospital, 55 Fruit Street, Boston, MA, 02114, USA.
⁵ Department of Psychiatry, Massachusetts General Hospital, One Bowdoin Square, 7th Floor, Boston, MA, 02114, USA. mpulsifer@mgh.harvard.edu.
⁶ Harvard Medical School, Boston, MA, USA. mpulsifer@mgh.harvard.edu.

Abstract

Purpose: Post-operative pediatric cerebellar mutism syndrome (CMS), characterized by mutism, ataxia/hypotonia, and emotional lability, can result in long-term deficits following resection of posterior fossa (PF) tumors. This longitudinal study compared neuropsychological outcomes of pediatric patients with post-operative CMS to a matched control patient group without CMS.

Methods: Fifty-eight PF tumor patients received post-surgical proton radiation therapy (PRT) and testing at baseline and at ≥ 1-year post-PRT over a 10-year period. Of these, 18 (31%) had post-operative CMS with baseline and follow-up neuropsychological test data. Those participants were matched to 18 controls by tumor location, age, gender, and handedness; no significant group differences were found at baseline for clinical/demographic variables. Total mean age at baseline was 7.26 years (SD = 4.42); mean follow-up interval was 3.26 years (SD = 2.24). Areas assessed: overall intelligence, expressive and receptive vocabulary, visuomotor integration, fine motor speed, inhibition, emotional control, depression, and anxiety.

Results: Patients were 52% male; 86% medulloblastoma/14% ependymoma; 86% craniospinal irradiation/14% focal radiation; and 86% chemotherapy. No group differences were found between most mean baseline scores; expressive vocabulary and fine motor speed were significantly lower in the post-operative CMS group (p < 0.05). Mean change scores revealed no significant differences for the sample; scores were within the normal range except fine motor skills were impaired for both groups.

Conclusions: Longitudinal neuropsychological outcomes for post-operative pediatric CMS patients did not differ significantly from matched controls without this condition. Patients were in the normal range in all areas except fine motor speed, which was impaired for both groups independent of CMS diagnosis.

Keywords: Brain tumor; Neuropsychological outcomes; Post-operative pediatric cerebellar mutism syndrome; Posterior fossa syndrome; Proton radiation therapy.

Publication types

Research Support, N.I.H., Extramural

MeSH terms

Cerebellar Neoplasms* / radiotherapy
Cerebellar Neoplasms* / surgery
Child
Female
Humans
Longitudinal Studies
Male
Medulloblastoma* / radiotherapy
Medulloblastoma* / surgery
Mutism* / etiology
Neuropsychological Tests
Protons

Substances

Protons

Abstract

Publication types

MeSH terms

Substances

Grants and funding