Marginal Decrease in Mortality and Marked Increase in Incidence as a Result of Neuroblastoma Screening at 6 Months of Age: Cohort Study in Seven Prefectures in Japan

Abstract

PURPOSE: To determine the usefulness of 6-month screening for neuroblastoma. PATIENTS AND METHODS: The cumulative incidence rates (IRs) and cumulative mortality rates (MRs) of neuroblastoma in children younger than 60 months of age were analyzed for control (n = 713,025), qualitative screening (Qual Screen, n = 1,142,519), and quantitative screening (Quan Screen, n = 550,331) cohorts, and for Screened and Unscreened subgroups within screening cohorts. RESULTS: IRs (per 100,000) for infants aged 6 to 11 months were 1.12 in Control, 5.69 in Qual Screen (P < .0001), and 17.81 in Quan Screen (P < .0001); IRs for children aged 12 to 59 months were 7.29 in Control, 5.86 in Qual Screen (P = .28), and 6.36 in Quan Screen (P = .60). IRs for children aged 12 to 59 months in Unscreened or Screened subgroups remained at the same level. When patients diagnosed at younger than 6 months of age were excluded, the MR (per 100,000) under 60 months for Control was 4.21; those in Unscreened and Screened subgroups were 3.84 and 2.53 in Qual Screen (P = .30), and 3.20 and 1.97 in Quan Screen (P = .73), respectively; MRs between Control and Unscreened subgroups revealed no significant differences (P = .89 in Qual Screen, P = .85 in Quan Screen). CONCLUSION: Six-month screening resulted in a marked increase in incidence for infants with no significant decrease in incidence for children older than 1 year of age. A decrease in mortality was observed, but it was not significant. The usefulness of screening is questionable, because the decrease of mortality should be balanced against the adverse effect of overdiagnosis and the psychological burden on parents and children.