Premorbid functioning as a predictor of outcome in pediatric brain tumor: An initial examination of the normalcy assumption

Pediatr Blood Cancer. 2020 Apr;67(4):e28135. doi: 10.1002/pbc.28135. Epub 2019 Dec 30.

Abstract

Background: Research on neurodevelopmental outcome in survivors of pediatric brain tumor (BT) is often based on the assumption of normal development up to the onset of overt symptoms. We sought to verify the "normalcy assumption" and to investigate corollary issues including challenges inherent to the measurement of premorbid neurobehavioral functioning.

Procedure: The Brain Radiation Investigative Study Consortium (BRISC) is a prospective longitudinal multisite study of 58 children diagnosed with BT. Premorbid functioning was assessed via retrospective parent report on standardized rating scales and detailed questionnaires. Findings were examined for the sample as a whole and in patients grouped by tumor histology (embryonal and non-embryonal).

Results: Mean age at diagnosis was 9.84 years (range, 3-16). The overall sample showed low proportions of pre/postnatal risk factors and delays in development. The proportion of children with clinically significant premorbid attention (18%) problems based on the BASC-2 exceeded expectation of that in healthy children (6.68%). Similar findings were obtained for somatization (18%) and anxiety (14%). Delays in talking were significantly more common in children with embryonal than non-embryonal tumors (P = 0.02). The non-embryonal tumor group had significantly higher overall rates of premorbid psychosocial problems than the embryonal tumor group (P < 0.001).

Conclusions: We describe a rigorous approach to estimating premorbid developmental status in pediatric BT. The findings suggest mixed support for the "normalcy assumption" and highlight the complexity of this concept and need for further investigation. Our results also suggest the need for further study of potential premorbid correlates with tumor histology.

Keywords: BASC-2; attention; language development; pre/postnatal; risk factor.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Adolescent
  • Brain Neoplasms / complications*
  • Brain Neoplasms / pathology
  • Child
  • Child Behavior Disorders / complications*
  • Child, Preschool
  • Developmental Disabilities / complications*
  • Female
  • Humans
  • Male
  • Retrospective Studies
  • Risk Factors