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Screening for neuroblastoma in France: methodological aspects and preliminary observations
F Chauvin1, P Mathieu, D Frappaz
1Service de Biologie Générale et de Neurobiologie, C.H.S. le Vinatier, Bron, France.
Insights
This pilot study in France demonstrates the feasibility of mass neuroblastoma screening. High-pressure liquid chromatography (HPLC) proved effective, with high participation rates and identification of good-prognosis tumors.
Area of Science:
- Pediatric Oncology
- Public Health Screening
- Biochemical Analysis
Background:
- Neuroblastoma is a significant childhood cancer.
- Early detection through mass screening can improve outcomes.
- A pilot study was conducted in the Rhône French district to assess screening feasibility.
Purpose of the Study:
- To measure compliance rates for a voluntary neuroblastoma screening test.
- To evaluate high-pressure liquid chromatography (HPLC) as a screening method.
- To characterize detected neuroblastoma tumors biologically.
Main Methods:
- A 5-year pilot study involving mass screening of newborns.
- Voluntary testing at 4 months of age using high-pressure liquid chromatography (HPLC).
- Data collection on participation rates, test accuracy, and tumor characteristics.
Main Results:
- Screening compliance increased from 69% to over 83% during the study period.
- High-pressure liquid chromatography (HPLC) demonstrated satisfactory assay performance with low false positive rates.
- Eight neuroblastomas were detected via screening, all with good prognoses; two false negatives and three missed cases due to noncompliance were identified.
Conclusions:
- Mass screening for neuroblastoma is feasible in France.
- The study highlights the potential for early detection of good-prognosis neuroblastomas.
- Consideration of overdiagnosis and the impact of noncompliance are crucial for program design.
Abstract:
A pilot study of neuroblastoma mass screening was initiated in January 1990 in the Rhône French district. The expected number of births per year is 26,000. The study is designed for a 5-year period with three major goals: 1) measurement of the compliance rate of a voluntary test at 4 months of age; 2) evaluation of the technical value of high-pressure liquid chromatography (HPLC) as a screening method; and 3) detailed biological characterization of all detected tumors. 61,551 children were screened between May 1, 1990 and December 31, 1993. Participation was 69% in 1990, 81.5% in 1991, and over 83% in 1992. HPLC was a satisfactory assay method. The number of clinical examinations required for positive tests as defined in the protocol is 1 per 3,621 tests. The false positive rate is 1 per 3,583 tests. Eight neuroblastomas were discovered by-screening (one stage I, three stage II, one stage III, three stage IVs). All are alive and well but were good prognosis cases according to the main prognostic factors. Five patients were discovered before screening (so called Halo effect): one stage I, one stage III, three stage IVs. One died of disease and four are alive in complete remission after treatment. Two patients were false negative (one stage III with N-myc amplification, one stage IV with bad prognosis features) and three cases of neuroblastoma were missed because of noncompliance with the screening program. This pilot study concludes on the feasibility of a mass screening program in France. The estimated cumulative incidence of neuroblastoma at 3 years is 1 per 4,375 living births and overdiagnosis is probable. All the detected cases were of good prognosis and the false negative ones were poor prognosis cases.