Incidence and survival among children with neuroblastoma in Spain over 22 years.
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BACKGROUND: Neuroblastoma is the most common extracranial solid childhood tumour. Advances in risk stratification have improved survival by enabling better-tailored treatments. The Toronto Guidelines (TG) aim to standardise childhood cancer stage collection by cancer registries. However, population-based survival analyses by stage remain scarce. We aimed to study incidence and survival of neuroblastoma cases among Spanish children aged < 15 years, with special attention to the role of stage at diagnosis. METHODS: The Spanish Registry of Childhood Tumours (RETI-SEHOP) and the Spanish Neuroblastoma Clinical Database (NBL-CDB) were cross-linked to achieve nearly complete registration of neuroblastoma in Spain for the period of diagnosis 2000–2021. Kaplan-Meier estimates were obtained for overall survival (OS). Adjusted survival was estimated using a Cox proportional hazards model with stratification by age group. RESULTS: During 2000–2021, 1774 neuroblastoma cases aged 0–14 years were recorded, making a rough average of 81 cases diagnosed annually. Age-standardised incidence rate (World) 2000–2021: 14.0 cases per million children (0–14 years). Distribution of stage at diagnosis remained stable from 2000 to 2021. 5-year OS increased from 74% (2000–2005) to 81% (2012–2017). Statistically significant OS differences were identified between sexes, age groups, tumour morphologies, primary sites and stages. Over time, survival improvement was centred mostly on male and locoregional cases. CONCLUSIONS: Neuroblastoma incidence and survival results in Spain were consistent with those from other high-income countries. Survival improvements were related to changes in clinical management, such as administration of anti-GD2 immunotherapy or the start of the LINES clinical trial (EudraCT 2010-021396-81, registered on 15 June 2011; ClinicalTrials.gov identifier: NCT01728155). This study underscores the clinical interest in stage data being collected by cancer registries, as per the TG.