AUTHOR=Lopez-Cortes Angela , Didonè Fabio , Botta Laura , Hjalgrim Lisa L. , Jakab Zsuzsanna , Cañete Nieto Adela , Stiller Charles , Zeller Bernward , Gatta Gemma , Pritchard-Jones Kathy , The BENCHISTA Project Working Group , Aitken Joanne , O’Neil Leisa , Hack Monika , Ladenstein Ruth , Van Eycken Elizabeth , Van Damme Nancy , Frazier Lindsay , De Camargo Beatriz , de Oliveira Santos Marceli , Valerianova Zdravka , Konstantinov Dobrin , Gupta Sumit , D Pole Jason , Sekerija Mario , Stary Jan , Sterba Jaroslav , Hjalgrim Lisa L. , Winther Jeanette F , Paapsi Keiu , Lacour Brigitte , Desandes Emmanuel , Clavel Jacqueline , Poulalhon Claire , Erdmann Friederike , Petridou Eleni T , Bouka Evdoxia , Jakab Zsuzsanna , Mian Michael , Galasso Rocco , Sampietro Giuseppe , Vetrano Francesco , Maule Milena M , Sacerdote Carlotta , Ballotari Paola , De Santis Emilia , Ferrante Margherita , Ragusa Rosalia , Boni Luca , Rognoni Magda , Amodio Rosalba , Boschetti Lorenza , Cuccaro Francesco , Bruno Danila , Russo Antonio G , Gervasi Federico , Gambino Maria L , Borciani Elisabetta , Michiara Maria , Mangone Lucia , Spagnoli Gianbattista , Ferretti Stefano , Falcini Fabio , Spata Eugenia , Manasse Sonia , Coccia Paola , Bella Francesca , Caldarella Adele , Intrieri Teresa , Scuderi Tiziana , Rizzello Roberto V , Rugge Massimo , Guzzinati Stefano , Murray Deirdre , Matsuda Tomohiro , Nakata Kayo , Azzopardi Miriam J , Dahlen Aina H , Tom Børge Johannesen , Zeller Bernward , Kowalczyk Jerzy , Jedrzejczyk Monika , Caldas Gabriela , Bucurenci Mihaela , Coza Dana , Zadnik Vesna , Lopez de Munain Arantza , Almela-Vich Fernando , Fuster-Camarena Nieves , Marcos-Gragera Ra f a e l , Sanchez Maria José , Aragones Nuria , Lopez Raquel , Dolores Chirlaque Maria , Guevara Marcela , Pardo Elena , Peris-Bonet Rafael , Cañete Nieto Adela , Carulla Marià , Lähteenmäki Päivi , Kuehni Claudia E , Redmond Shelagh M , Karim-Kos Henrike , Stiller Charles , Stacey Paul , Irvine Lucy , Gavin Anna , Fitzpatrick Deirdre , Morrison David S , Smith Karen , Huws Dyfed Wyn , Warlow Janet , Strauss Sandra , Bailey Simon , Nieto Adela Canete , Gaspar Nathalie , Hjalgrim Lisa L. , Spreafico Filippo , Polanco Angela , Capocaccia Riccardo , Di Cataldo Andrea , Klein Meric TITLE=Cancer data quality and harmonization in Europe: the experience of the BENCHISTA Project – international benchmarking of childhood cancer survival by stage JOURNAL=Frontiers in Oncology VOLUME=13 YEAR=2023 URL=https://www.frontiersin.org/journals/oncology/articles/10.3389/fonc.2023.1232451 DOI=10.3389/fonc.2023.1232451 ISSN=2234-943X ABSTRACT=Introduction

Variation 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.

Methods

PBCRs 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.

Results

67 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)].

Conclusion

Differences 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.