Sporadic unilateral retinoblastoma or first sign of bilateral disease?

Br J Ophthalmol. 2013 Apr;97(4):475-80. doi: 10.1136/bjophthalmol-2012-302666. Epub 2013 Jan 26.

Abstract

Background: A small number of children with unilateral retinoblastoma later develop retinoblastoma in the contralateral eye (metachronous bilateral retinoblastoma).

Methods: We analysed the clinical and genetic characteristics of children with sporadic unilateral retinoblastoma to identify risk factors for the development of metachronous bilateral disease.

Results: Fifteen (3.1%) of 480 children with unilateral retinoblastoma later developed metachronous bilateral retinoblastoma (latency period >30 days). The maximum latency period was 2.3 years after initial diagnosis. Nine (22.5%) of 40 children with a RB1 mutation detectable in blood developed metachronous bilateral disease while all 155 children proved to be without a germline RB1 mutation remained unilaterally affected. Clinically, the risk of developing metachronous bilateral retinoblastoma was higher for age at diagnosis ≤0.5 years compared with >0.5 years (19.6% vs 1.2%), and for multifocal compared with unifocal unilateral retinoblastoma (17.1% vs 2.2%).

Conclusions: This study shows that an oncogenic RB1 mutation in the blood is a risk factor for metachronous bilateral retinoblastoma. Additional clinical risk factors for metachronous bilateral disease are diagnosis at young age (≤0.5 years) and multifocal unilateral retinoblastoma. Early genetic analysis may identify children at high risk of developing metachronous bilateral disease and may help to preserve vision using risk-adapted follow-up and early treatment.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age Distribution
  • Child
  • Child, Preschool
  • DNA Mutational Analysis
  • Follow-Up Studies
  • Humans
  • Infant
  • Infant, Newborn
  • Neoplasms, Second Primary / diagnosis*
  • Neoplasms, Second Primary / genetics
  • Real-Time Polymerase Chain Reaction
  • Retinal Neoplasms / diagnosis*
  • Retinal Neoplasms / genetics
  • Retinoblastoma / diagnosis*
  • Retinoblastoma / genetics
  • Retinoblastoma Protein / genetics
  • Retrospective Studies
  • Risk Factors

Substances

  • Retinoblastoma Protein