Cancer data quality and harmonization in Europe: the experience of the BENCHISTA Project – international benchmarking of childhood cancer survival by stage

Frontiers in Oncology

Oncology

Lisa L. Hjalgrim¹  Adela Canete Nieto¹  Zsuzsanna Jakab¹  Charles Stiller¹  Bernward Zeller¹  Laura Botta²  Gemma Gatta²  Fabio Didonè²  Angela Lopez-Cortes³  Kathy Pritchard-Jones³ 

[1] BENCHISTA Project Management Team, London, United Kingdom;Fondazione IRCCS “Istituto Nazionale dei Tumori di Milano” (INT), Department of Evaluative Epidemiology, Milan, Italy;University College London (UCL) Great Ormond Street Institute of Child Health, Developmental Biology & Cancer Research Department, London, United Kingdom;

关键词: childhood cancer;    population-based;    cancer registry;    Toronto staging;    diagnosis;    survival;    data quality;    data harmonization;   

DOI  :  10.3389/fonc.2023.1232451

 received in 2023-05-31, accepted in 2023-07-24,  发布年份 2023

来源: Frontiers

【 摘 要 】

IntroductionVariation in stage at diagnosis of childhood cancers (CC) may explain differences in survival rates observed across geographical regions. The BENCHISTA project aims to understand these differences and to encourage the application of the Toronto Staging Guidelines (TG) by Population-Based Cancer Registries (PBCRs) to the most common solid paediatric cancers.MethodsPBCRs within and outside Europe were invited to participate and identify all cases of Neuroblastoma, Wilms Tumour, Medulloblastoma, Ewing Sarcoma, Rhabdomyosarcoma and Osteosarcoma diagnosed in a consecutive three-year period (2014-2017) and apply TG at diagnosis. Other non-stage prognostic factors, treatment, progression/recurrence, and cause of death information were collected as optional variables. A minimum of three-year follow-up was required. To standardise TG application by PBCRs, on-line workshops led by six tumour-specific clinical experts were held. To understand the role of data availability and quality, a survey focused on data collection/sharing processes and a quality assurance exercise were generated. To support data harmonization and query resolution a dedicated email and a question-and-answers bank were created.Results67 PBCRs from 28 countries participated and provided a maximally de-personalized, patient-level dataset. For 26 PBCRs, data format and ethical approval obtained by the two sponsoring institutions (UCL and INT) was sufficient for data sharing. 41 participating PBCRs required a Data Transfer Agreement (DTA) to comply with data protection regulations. Due to heterogeneity found in legal aspects, 18 months were spent on finalizing the DTA. The data collection survey was answered by 68 respondents from 63 PBCRs; 44% of them confirmed the ability to re-consult a clinician in cases where stage ascertainment was difficult/uncertain. Of the total participating PBCRs, 75% completed the staging quality assurance exercise, with a median correct answer proportion of 92% [range: 70% (rhabdomyosarcoma) to 100% (Wilms tumour)].ConclusionDifferences in interpretation and processes required to harmonize general data protection regulations across countries were encountered causing delays in data transfer. Despite challenges, the BENCHISTA Project has established a large collaboration between PBCRs and clinicians to collect detailed and standardised TG at a population-level enhancing the understanding of the reasons for variation in overall survival rates for CC, stimulate research and improve national/regional child health plans.

【 授权许可】

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Copyright © 2023 Lopez-Cortes, Didonè, Botta, Hjalgrim, Jakab, Canete Nieto, Stiller, Zeller, Gatta, Pritchard-Jones and The BENCHISTA Project Working Group