NEUROBLASTOMA SCREENING IN INFANTS POSTPONED AFTER THE 6TH-MONTH OF AGE - A TRIAL TO REDUCE OVERDIAGNOSIS AND TO DETECT CASES WITH UNFAVORABLE BIOLOGIC FEATURES
R. Kerbl et al., NEUROBLASTOMA SCREENING IN INFANTS POSTPONED AFTER THE 6TH-MONTH OF AGE - A TRIAL TO REDUCE OVERDIAGNOSIS AND TO DETECT CASES WITH UNFAVORABLE BIOLOGIC FEATURES, Medical and pediatric oncology, 29(1), 1997, pp. 1-10
Background. Encouraged by Japanese reports of the benefits of screenin
g 6-month-old infants for neuroblastoma, a neuroblastoma screening pro
gram was introduced in Austria in 1991. However, because of concerns r
elated to ''overdiagnosis'' by screening at this age, the screening te
st was performed at a later age. Methods. From March 1991 to February
1995 neuroblastoma screening was performed on filter paper urine speci
mens in 100,043 Austrian infants (median age 8.5 months). Primary anal
ysis of urine catecholamines (vanillylmandelic acid and homovanillic a
cid) was per formed by use of an EIA method. Questionable or positive
results were confirmed by high performance liquid chromatography (HPLC
). A double retest was requested following a positive HPLC result. Res
ults. Twenty-one infants were admitted to a hospital following repeate
dly elevated values of vanillylmandelic acid (VMA) and/or homovanillic
acid (HVA). Eleven infants were found to have neuroblastoma (three st
age 1, four stage 2 B, lour stage 3). Treatment consisted of surgery a
lone with total or subtotal resection in eight cases, surgery and chem
otherapy in two cases, and chemotherapy alone in one case. Biologic fe
atures were assessed in all tumors excluding ploidy in one case. The m
ajority of the tumors analyzed were near-triploid (9/10), however, two
tumors revealed N-myc amplification. Conclusion. Our results demonstr
ate that stage distribution and biologic features oi neuroblastomas di
agnosed by screening at 8.5 months are different from the results of s
creening at 6 months. Furthermore, the detection of one neuroblastoma
among 9,100 screened infants is significantly lower than the incidence
of the Japanese screening program. Our results suggest that screening
at an age of 7 to 10 months reduces overdiagnosis and may be of more
benefit than earlier screening. (C) 1997 Wiley-Liss, Inc.