Prognostic factors of overall survival in children and adolescents enrolled in dose-finding trials in Europe: An Innovative Therapies for Children with Cancer study

Eur J Cancer. 2016 Nov:67:130-140. doi: 10.1016/j.ejca.2016.08.008. Epub 2016 Sep 21.

Abstract

Objectives: Dose-finding trials are fundamental to develop novel drugs for children and adolescents with advanced cancer. It is crucial to maximise individual benefit, whilst ensuring adequate assessment of key study end-points. We assessed prognostic factors of survival in paediatric phase I trials, including two predictive scores validated in adult oncology: the Royal Marsden Hospital (RMH) and the MD Anderson Cancer Center (MDACC) scores.

Methods: Data of patients with solid tumours aged <18 years at enrolment in their first dose-finding trial between 2000 and 2014 at eight centres of the Innovative Therapies for Children with Cancer European consortium were collected. Survival distributions were compared using log-rank test and Cox regression analyses.

Results: Overall, 248 patients were evaluated: median age, 11.2 years (range 1.0-17.9); 46% had central nervous system (CNS) tumours and 54% extra-CNS tumours. Complete responses were observed in 2.1%, partial responses in 7.2% and stable disease in 25.9%. Median overall survival (OS) was 6.3 months (95% confidence interval, 5.2-7.4). Lansky/Karnofsky ≤80%, no school/work attendance, elevated creatinine and RMH score ≥1 correlated with worse OS in the multivariate analysis. The RMH and MDACC scores correlated with OS in adolescents (12-17 years), p = 0.002, but not in children (2-11 years).

Conclusions: Performance status of 90-100% and school/work attendance at enrolment are strong indicators of longer OS in paediatric phase I trials. Adult predictive scores correlate with survival in adolescents. These findings provide a useful orientation about potential prognosis and could lead in the future to more paediatric-adapted eligibility criteria in early-phase trials.

Keywords: Adolescents; Children; Dose-finding trial; Innovative Therapies for Children with Cancer; Phase I trial; Prognostic factor; Survival.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Central Nervous System Neoplasms / drug therapy
  • Central Nervous System Neoplasms / mortality
  • Child
  • Child, Preschool
  • Clinical Trials, Phase I as Topic
  • Employment*
  • Ependymoma / drug therapy
  • Ependymoma / mortality
  • Europe
  • Female
  • Glioma / drug therapy
  • Glioma / mortality
  • Humans
  • Infant
  • Male
  • Medulloblastoma / drug therapy
  • Medulloblastoma / mortality
  • Multivariate Analysis
  • Neoplasms / drug therapy
  • Neoplasms / mortality*
  • Neuroblastoma / drug therapy
  • Neuroblastoma / mortality
  • Neuroectodermal Tumors, Primitive / drug therapy
  • Neuroectodermal Tumors, Primitive / mortality
  • Osteosarcoma / drug therapy
  • Osteosarcoma / mortality
  • Prognosis
  • Proportional Hazards Models
  • Retrospective Studies
  • Rhabdomyosarcoma / drug therapy
  • Rhabdomyosarcoma / mortality
  • Sarcoma / drug therapy
  • Sarcoma / mortality
  • Sarcoma, Ewing / drug therapy
  • Sarcoma, Ewing / mortality
  • Schools*
  • Survival Rate
  • Therapies, Investigational