Is neuroblastoma screening evaluation needed and feasible?

Abstract

Despite the five million children who have been screened for neuroblastoma in Japan through detection of catecholamine metabolites, it is still uncertain whether screening for this disease is beneficial. The Japanese study has clearly indicated that screening at 6 months or earlier leads to heavy overdiagnosis. It is shown in this paper that screening at a later age may give the same reduction in mortality with possibly less overdiagnosis. However, it is estimated that, even with two screens at 12 and 18 months, the reduction in mortality would not greatly exceed 25%, under realistic hypotheses on the length of the preclinical phase of the disease. The evaluation of the efficacy of this screening strategy would need the recruitment of half a million children per year over 5-7 years and the follow-up of an equal number of controls. Such a trial would improve our knowledge of the natural history of the disease and might help to answer some questions raised recently regarding its biological heterogeneity.

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

“…Whereas some Japanese reports describe a decrease in the incidence of cases of advanced neuroblastoma in older children after the introduction of neuroblastoma screening [4][5][6], other authors doubt about the efficiency of screening at 6 months [17][18][19][20]22,34]. The question whether screening at 6 months of age can reduce mortality from neuroblastoma has not yet been answered with statistical significance and requires a huge controlled study [35,36]. However, a significant increase of neuroblastoma incidence in areas with early screening (6 months or earlier) probably means that many infants are treated unnecessarily.…”

“…Due to the population size, even with a 100% compliance rate a controlled study (50% screened, 50% unscreened) would take about 30 years to answer epidemiological questions concerning mortality with statistical significance [36]. Therefore, at the moment the Austrian study is continued as a ''pilot study'' with the aim to detect further neuroblastoma cases with unfavorable biologic features at an early clinical stage.…”

“…Some authors argue that neuroblastoma mortality may be little affected by early screening [15,34]. Thus, the need for prospective controlled studies was emphasized [35], but these studies require huge numbers of screened and unscreened infants [36].…”

Editorial: objective approach of the screening question

“…Overdiagnosis also risks treatment-related morbidity and mortality if surgery is done on all detected cases. The experience of all studies at 6 months or earlier is that screening at this age is inadvisable [5].…”

Diagnostic imaging in abdominal neuroblastoma: Is there a complementary role of MIBG-scintigraphy and ultrasonography?

“…It is still controversial whether mass screening in infants can improve the prognosis of the disease. Being supported by some [13,22,33], (especially early) neuroblastoma screening faces scepticism by others [5,6,10,14] due to the embryonic origin of the tumour paralleling with heterogeneous biologic features and clinical behaviour.…”