Neuroblastoma screening in infants postponed after the sixth month of age: a trial to reduce "overdiagnosis" and to detect cases with "unfavorable" biologic features.
BACKGROUND
Encouraged by Japanese reports of the benefits of screening 6 month-old infants for neuroblastoma, a neuroblastoma screening program was introduced in Austria in 1991. However, because of concerns related to "overdiagnosis" by screening at this age, the screening test was performed at a later age.


METHODS
From March 1991 to February 1995 neuroblastoma screening was performed on filter paper urine specimens in 100,043 Austrian infants (median age 8.5 months). Primary analysis of urine catecholamines (vanillylmandelic acid and homovanillic acid was performed by use of an E1A method. Questionable or positive results were confirmed by high performance liquid chromatography (HPLC). A double retest was requested following a positive HPLC result.


RESULTS
Twenty-one infants were admitted to a hospital following repeatedly elevated values of vanillymandelic acid (VMA) and/or homovanillic acid (HVA). Eleven infants were found to have neuroblastoma (three stage 1, four stage 2B, four stage 3). Treatment consisted of surgery alone with total or subtotal resection in eight cases, surgery and chemotherapy in two cases, and chemotherapy alone in one case. Biologic features were assessed in all tumors excluding ploidy in one case. The majority of the tumors analyzed were near-triploid (9/10), however, two tumors revealed N-myc amplification.


CONCLUSIONS
Our results demonstrate that stage distribution and biologic features of neuroblastomas diagnosed by screening at 8.5 months are different from the results of screening 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.
Kerbl
R
R
University Children's Hospital (Division of Hematology/Oncology), Graz, Austria.
Urban
C E
CE
Ladenstein
R
R
Ambros
I M
IM
Spuller
E
E
Mutz
I
I
Amann
G
G
Kovar
H
H
Gadner
H
H
Ambros
P F
PF
D000367:Age Factors; D001317:Austria / Q000453:epidemiology; D006801:Humans; D015994:Incidence; D007223:Infant; D008403:Mass Screening; D009367:Neoplasm Staging; D009447:Neuroblastoma / Q000175:diagnosis* / Q000453:epidemiology / Q000473:pathology / Q000517:prevention & control