Developmental and adaptive functioning in children with retinoblastoma: a longitudinal investigation

J Clin Oncol. 2014 Sep 1;32(25):2788-93. doi: 10.1200/JCO.2013.53.1996. Epub 2014 Jul 14.

Abstract

Purpose: To determine the developmental trajectory of early cognitive and adaptive skills in young children with retinoblastoma from diagnosis to 5 years of age.

Patients and methods: Ninety-four patients with retinoblastoma treated according to an institutional protocol underwent serial assessments of cognitive and adaptive functioning at age 6 months and 1, 2, 3, and 5 years. Data were analyzed by treatment strata, with patients with 13q deletion analyzed separately.

Results: At baseline, across all patients (except those with 13q deletion), developmental functioning was comparable with the normative mean, with mean scores for all strata within the average range. However, at age 5 years, developmental functioning was in the low average range and significantly below normative means. The trajectories of developmental functioning demonstrated significant decline over time, although this varied by treatment group/strata. Patients treated with enucleation only evidenced the greatest decline in cognitive functioning; significant change was not observed in patients treated with other modalities. Notable declines in parent-reported communication skills were observed in the majority of patients. Patients with 13q deletion evidenced delayed cognitive functioning at baseline, but minimal declines were observed through age 3 years. However, significant decreases in adaptive functioning were demonstrated over time for the 13q deletion subset.

Conclusion: The declines in functioning observed in this study were unexpected, as was the poorer performance of the enucleation-only group. This highlights the necessity of continuing to assess cognitive functioning in patients with retinoblastoma as they age. Additional research is necessary to determine the long-term trajectory of cognitive development in this population.

MeSH terms

  • Adaptation, Physiological / physiology*
  • Child
  • Child, Preschool
  • Cognition / physiology
  • Female
  • Humans
  • Infant
  • Infant, Newborn
  • Longitudinal Studies
  • Male
  • Retinal Neoplasms / physiopathology*
  • Retinal Neoplasms / psychology
  • Retinoblastoma / physiopathology*
  • Retinoblastoma / psychology