Neurocognitive, academic and functional outcomes in survivors of infant ependymoma (UKCCSG CNS 9204)

Childs Nerv Syst. 2019 Mar;35(3):411-420. doi: 10.1007/s00381-018-4015-3. Epub 2018 Dec 15.

Abstract

Purpose: This is the first UK multi-centre case-controlled study with follow-up in excess of 10 years to report the neurocognitive, academic and psychological outcomes of individuals diagnosed with a brain tumour in early childhood. Children enrolled into the UKCCSG CNS 9204 trial, diagnosed with intracranial ependymoma when aged ≤ 36 months old, who received a primary chemotherapy strategy to defer or avoid radiotherapy, were recruited.

Methods: Outcomes of those who relapsed and subsequently received radiotherapy (n = 13) were compared to those enrolled who did not relapse (n = 16), age-matched controls-diagnosed with solid non-central nervous system (SN-CNS; n = 15) tumours or low-grade posterior fossa pilocytic astrocytoma (PFPA; n = 15), and normative data. Analyses compared nine neurocognitive outcomes as primary measures with quality of survival as secondary measures.

Results: Relapsed ependymoma participants performed significantly worse than their non-relapsed counterparts on measures of Full Scale IQ, Perceptual Reasoning, Word Reading and Numerical Operations. The relapsed ependymoma group performed significantly worse than SN-CNS controls on all primary measures, whereas non-relapsing participants only differed significantly from SN-CNS controls on measures of Processing Speed and General Memory. Relapsed ependymoma participants fared worse than all groups on measures of quality of survival.

Conclusions: The relapsed irradiated ependymoma group demonstrated the most significantly impaired neurocognitive outcomes at long-term follow-up. Non-relapsing participants demonstrated better outcomes than those who relapsed. Results tentatively suggest avoiding radiotherapy helped preserve neurocognitive and learning outcomes of individuals diagnosed with ependymoma when aged ≤ 36 months old. Prospective neurocognitive surveillance is required. Recommendations for clinical and research practice are provided.

Keywords: Brain tumour; Ependymoma; Neurocognitive; Outcome; Paediatric; Quality of survival.

MeSH terms

  • Antineoplastic Combined Chemotherapy Protocols / therapeutic use
  • Brain Neoplasms / therapy*
  • Cancer Survivors / statistics & numerical data*
  • Case-Control Studies
  • Child Development / drug effects
  • Child Development / radiation effects
  • Child, Preschool
  • Ependymoma / therapy*
  • Female
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Neoplasm Recurrence, Local / radiotherapy
  • Neurocognitive Disorders / etiology*
  • Radiotherapy / adverse effects
  • Radiotherapy / methods
  • Recovery of Function / drug effects
  • Recovery of Function / radiation effects
  • United Kingdom

Supplementary concepts

  • Familial ependymoma