Integrating Tenascin-C protein expression and 1q25 copy number status in pediatric intracranial ependymoma prognostication: A new model for risk stratification

PLoS One. 2017 Jun 15;12(6):e0178351. doi: 10.1371/journal.pone.0178351. eCollection 2017.

Abstract

Purpose: Despite multimodal therapy, prognosis of pediatric intracranial ependymomas remains poor with a 5-year survival rate below 70% and frequent late deaths.

Experimental design: This multicentric European study evaluated putative prognostic biomarkers. Tenascin-C (TNC) immunohistochemical expression and copy number status of 1q25 were retained for a pooled analysis of 5 independent cohorts. The prognostic value of TNC and 1q25 on the overall survival (OS) was assessed using a Cox model adjusted to age at diagnosis, tumor location, WHO grade, extent of resection, radiotherapy and stratified by cohort. Stratification on a predictor that did not satisfy the proportional hazards assumption was considered. Model performance was evaluated and an internal-external cross validation was performed.

Results: Among complete cases with 5-year median follow-up (n = 470; 131 deaths), TNC and 1q25 gain were significantly associated with age at diagnosis and posterior fossa tumor location. 1q25 status added independent prognostic value for death beyond the classical variables with a hazard ratio (HR) = 2.19 95%CI = [1.29; 3.76] (p = 0.004), while TNC prognostic relation was tumor location-dependent with HR = 2.19 95%CI = [1.29; 3.76] (p = 0.004) in posterior fossa and HR = 0.64 [0.28; 1.48] (p = 0.295) in supratentorial (interaction p value = 0.015). The derived prognostic score identified 3 different robust risk groups. The omission of upfront RT was not associated with OS for good and intermediate prognostic groups while the absence of upfront RT was negatively associated with OS in the poor risk group.

Conclusion: Integrated TNC expression and 1q25 status are useful to better stratify patients and to eventually adapt treatment regimens in pediatric intracranial ependymoma.

Publication types

  • Multicenter Study

MeSH terms

  • Age of Onset
  • Child
  • Child, Preschool
  • Chromosomes, Human, Pair 1 / genetics*
  • DNA Copy Number Variations*
  • Ependymoma / diagnosis*
  • Ependymoma / genetics
  • Ependymoma / metabolism
  • Female
  • Humans
  • Infant
  • Male
  • Prognosis
  • Survival Analysis
  • Tenascin / metabolism*

Substances

  • Tenascin

Grants and funding

This study had financial support from INCa/Cancéropôle Ile de France (FA), Société Française des Cancers de l'Enfant/Enfants et Santé (JG), Cancer Research-UK (in support of Children’s Cancer and Leukemia Group) (JPK and RG) and the following charities: L'Etoile de Martin, Léa Princesse Eternelle, Sarah Petite Princesse, Mécénat Carrefour (JG), German Childrens Cancer Aid (DKKS) (TP), the James Tudor and Joseph Foote Foundations (RG). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.