Neuroblastoma Screening at 1 Year of Age: the Final Results of a Controlled Trial
Menée à l'aide de données épidémiologiques et cliniques issues d'un programme de dépistage du neuroblastome chez les enfants âgés d'un an, cette étude évalue l'effet de ce programme sur l'incidence des neuroblastomes de stade avancé et la mortalité spécifique
Background : Neuroblastoma screening aimed to reduce neuroblastoma-related mortality. The controlled trial showed a non-reduction of stage 4 incidence and preliminary mortality data. This article presents epidemiological and clinical data 20 years after cessation of the screening program.
Methods : The patients detected in the screening area were compared with the clinically diagnosed patients of the control area, the pre-study, and the post-study cohorts. All statistical tests were 2-sided.
Results : The cumulative incidence for ages 1-6 years of the study birth cohorts (1994-1999) in the screening area was 13.4 cases per 100,000 births (95% confidence interval [CI] = 12.2-14.6) based on 61.2% screening participants and 38.8% non-participants. Screening participants had a cumulative incidence of 15.7 (95%-CI = 14.0-17.4) per 100,000 births. The cumulative incidence in the contemporary control cohort was 9.3 (95%-CI = 8.2-10.3) per 100,000 births, 7.6 (95%-CI = 6.8-8.4) in the pre-study cohort, and 8.1 (95%-CI = 7.4-8.9) in the post-study cohort 2000-2004 (P<0.001 each). The increased incidence in the screening cohort was restricted to stages 1-3, while stage 4 incidence was not reduced. The cumulative mortality for deaths within 10 years from diagnosis and per 100,000 births remained unchanged. Stage 4 patients who were detected by screening had better biologic characteristics and an improved outcome compared to not by screening detected stage 4 cases.
Conclusions : Neuroblastoma screening at one year of age reduced neither stage 4 incidence nor neuroblastoma mortality and was affected by overdiagnosis leading to unnecessary treatment. A few by screening-detected stage 4 cases represent a biologically interesting subgroup but do not change the recommendation to close the ‘catecholamine-based neuroblastoma screening book’.
JNCI Cancer Spectrum , article en libre accès, 2020