figshare
Browse
ionc_a_1672892_sm4627.docx (21.08 kB)

Neurologic disorders in long-term survivors of neuroblastoma – a population-based cohort study within the Adult Life after Childhood Cancer in Scandinavia (ALiCCS) research program

Download (21.08 kB)
journal contribution
posted on 2019-10-08, 09:56 authored by Filippa Nyboe Norsker, Catherine Rechnitzer, Elisabeth Wreford Andersen, Karen Markussen Linnet, Line Kenborg, Anna Sällfors Holmqvist, Laufey Tryggvadottir, Laura-Maria Madanat-Harjuoja, Ingrid Øra, Halldora K. Thorarinsdottir, Kim Vettenranta, Andrea Bautz, Henrik Schrøder, Henrik Hasle, Jeanette Falck Winther

Background: Neuroblastoma is the commonest extracranial solid tumor of childhood, yet rare, and with poor survival before 1990, especially for high-risk disease; thus, information on late effects is sparse. With great advances in cancer treatment, survival has reached 80% in the Nordic countries. The aim of the study was to investigate the risk of developing neurologic disorders after neuroblastoma.

Material and methods: Through population-based cancer registries of four Nordic countries we identified 654 5-year survivors of neuroblastoma (diagnosed 1959–2008) and 133,668 matched population comparisons. We grouped neurologic diagnoses from national hospital registries into 11 main diagnostic categories and 56 disease-specific sub-categories and calculated relative risks (RRs), absolute excess risks (AERs), cumulative incidence and mean cumulative count (MCC). Information on cancer treatment was available for 49% of survivors.

Results: A hospital contact for a neurologic disorder was observed in 181 survivors 5 years or more from cancer diagnosis with 59 expected, yielding a RR of 3.1 (95% CI 2.7–3.6) and an AER of 16 per 1,000 person-years (95% CI 12–19). The most frequent disorders included epilepsy, paralytic syndromes, diseases of the eyes and ears and hearing loss. The cumulative incidence of any neurologic disorder was 31% in survivors 20 years after cancer diagnosis with a MCC of 0.5 unique diagnoses. All risks were highest in survivors of high-risk neuroblastoma.

Conclusion: Neuroblastoma survivors represent a population with a high risk of developing neurologic disorders. Our results should contribute to improving health care planning and underscores the need for systematic follow-up care of this vulnerable group of survivors.

Funding

This work was supported by a grant from the Danish Cancer Society’s Scientific Committee [R56-A3210-12-S2] and by a grant from the Danish Childhood Cancer Foundation [2011-44]. Neither funder had any role in study design, data collection or analysis, the decision to publish, or preparation of the manuscript.

History