Frontiers in Oncology (Aug 2023)

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

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

DOI
https://doi.org/10.3389/fonc.2023.1232451
Journal volume & issue
Vol. 13

Abstract

Read online

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.

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