Screening for neuroblastoma is ineffective in reducing the incidence of unfavourable advanced stage disease in older children

W. G. Woods, M. Tuchman, L. L. Robison, M. Bernstein, J. M. Leclerc, L. C. Brisson, J. Brossard, G. Hill, J. Shuster, R. Luepker, T. Byrne, S. Weitzman, G. Bunin, B. Lemieux, G. M. Brodeur

Research output: Contribution to journalArticlepeer-review

24 Scopus citations

Abstract

Neuroblastoma exhibits many characteristics which would suggest that preclinical detection may improve outcome. The Quebec Neuroblastoma Screening Project was initiated to determine whether mass screening could reduce mortality in a large cohort of infants. All 476 603 children born in the province of Quebec during a 5-year period of time (1 May 1989 to 30 April 1994) were eligible for determinations of urinary catecholamine metabolites at 3 weeks and 6 months of age. Children with positive screening were referred to one of four paediatric cancer centres in Quebec for uniform evaluation and treatment. Standardised incidence ratios (SIRs) were calculated for neuroblastoma in Quebec and two comparable population-based controls during the same period of time using similar ascertainment procedures. Compliance with screening in Quebec was 91% at 3 weeks (n = 425 816) and 74% at 6 months (n = 349 706). Up to 31 July 1995 with a follow-up of the birth cohort of 15-75 months, 118 cases of neuroblastoma were diagnosed, 43 detected preclinically by screening, 20 detected clinically prior to screening at 3 weeks of age and 55 detected clinically after 3 weeks of age having normal screens (n = 52) or never screened (n = 3). Based on data from concurrent control populations, 54.5 cases of neuroblastoma would have been expected in Quebec during the study period for an SIR of 2.17 (95% CI 1.79-2.57, P< 0.0001). For the two control groups, the overall SIR was 1.00 (NS). SIRs for Quebec by age at diagnosis in yearly intervals show a marked increased incidence under I year of age (SIR = 2.85, 95% CI 2.26- 3.50), with no reduction in incidence in subsequent years. We conclude that screening for neuroblastoma markedly increases the incidence in infants without decreasing the incidence of unfavourable advanced stage disease in older children. It is unlikely that screening for neuroblastoma in infants will reduce the mortality of this disease.

Original languageEnglish (US)
Pages (from-to)2106-2112
Number of pages7
JournalEuropean Journal of Cancer
Volume33
Issue number12
DOIs
StatePublished - Oct 1997

Bibliographical note

Funding Information:
Supported in part by grants CA 46907 (WGW) and CA-39771 (GMB) from the United States National Institutes of Health, the Quebec Network for Genetic Medicine, Montreal, PQ and the National Cancer Institute of Canada (Grant #2691). Some of these data have been described previously [1] . Without the cooperation of the population of Quebec, this study would have been impossible. The authors thank the data managers at the four Quebec medical centres, Ms Aline Blais, Patricia Campion, Doreen Dupuis and Louise Renaud for their roles in gathering data and biological samples; and Ms Anne Feltis in Ontario who performed a similar role. Finally, the authors thank the project Oversite Committee consisting of John Potter, M.D., Ph.D., Harvey Levy, M.D., and Robert Castleberry, M.D., for many helpful criticisms and suggestions throughout the study.

Keywords

  • Cancer screening
  • Childhood cancer
  • Neuroblastoma
  • Newborn screening
  • Population studies

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