Neuroblastoma screening in infants postponed after the sixth month of age: A trial to reduce “overdiagnosis” and to detect cases with “unfavorable” biologic features

Publisher Website
Google Scholar
Abstract
Encouraged by Japanese reports of the benefits of screening 6-month-old infants of 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. 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 (vanillylmandolic acid and homovanillic acid) was performed 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. Twenty-one infants were admitted to a hospital following repeatedly elevated values of vanillylmandelic 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. 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 detections 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. Med. Pediatr. Oncol. 29:1–10, 1997.