Decision making for health‐related research outcomes that alter diagnosis: A model from paediatric brain tumours

Author:

Pickles Jessica C.12ORCID,Aquilina Kristian3,Chalker Jane4,Dahl Christine3,Devadass Abel5,Mankad Kshitij3,Merve Ashirwad2,Ahmed Munaza6,Nicoll James A. R.78ORCID,Bloom Tabitha7,Hilton David A.9,Sebire Neil J.12,Hargrave Darren13,Jacques Thomas S.12ORCID

Affiliation:

1. Developmental Biology and Cancer & Teaching Department UCL Great Ormond Street Institute of Child Health London UK

2. Department of Histopathology NIHR Great Ormond Street Hospital Biomedical Research Centre and UCL London UK

3. Department of Paediatric Haematology and Oncology Great Ormond Street Hospital for Children NHS Foundation Trust London UK

4. Specialist Integrated Haematology and Malignancy Diagnostic Service‐Acquired Genomics Great Ormond Street Hospital for Children NHS Foundation Trust London UK

5. Beaumont Hospital Dublin Ireland

6. North East Thames Regional Clinical Genetics Service Great Ormond Street Hospital for Children NHS Foundation Trust London UK

7. Clinical & Experimental Sciences University of Southampton Southampton UK

8. Cellular Pathology University Hospital Southampton NHS Foundation Trust Southampton UK

9. Department of Histopathology University Hospitals Plymouth Plymouth UK

Abstract

AbstractAimsThe question of how to handle clinically actionable outcomes from retrospective research studies is poorly explored. In neuropathology, this problem is exacerbated by ongoing refinement in tumour classification. We sought to establish a disclosure threshold for potential revised diagnoses as determined by the neuro‐oncology speciality.MethodsAs part of a previous research study, the diagnoses of 73 archival paediatric brain tumour samples were reclassified according to the WHO 2016 guidelines. To determine the disclosure threshold and clinical actionability of pathology‐related findings, we conducted a result‐evaluation approach within the ethical framework of BRAIN UK using a surrogate clinical multidisciplinary team (MDT) of neuro‐oncology specialists.ResultsThe MDT identified key determinants impacting decision‐making, including anticipated changes to patient management, time elapsed since initial diagnosis, likelihood of the patient being alive and absence of additional samples since cohort inception. Ultimately, none of our research findings were considered clinically actionable, largely due to the cohort's historic archival and high‐risk nature. From this experience, we developed a decision‐making framework to determine if research findings indicating a change in diagnosis require reporting to the relevant clinical teams.ConclusionsEthical issues relating to the use of archival tissue for research and the potential to identify actionable findings must be carefully considered. We have established a structured framework to assess the actionability of research data relating to patient diagnosis. While our specific findings are most applicable to the pathology of poor prognostic brain tumour groups in children, the model can be adapted to a range of disease settings, for example, other diseases where research is dependent on retrospective tissue cohorts, and research findings may have implications for patients and families, such as other tumour types, epilepsy‐related pathology, genetic disorders and degenerative diseases.

Funder

Brain Tumour Charity

Publisher

Wiley

Reference38 articles.

1. The 2021 WHO Classification of Tumors of the Central Nervous System: a summary

2. Cancer Research UK.Brain other CNS and intracranial tumours incidence statistics [Internet]. Accessed June 2022.https://www.cancerresearchuk.org/health-professional/cancer-statistics/statistics-by-cancer-type/brain-other-cns-and-intracranial-tumours/incidence#heading-One

3. BRAIN UK.BRAIN UK protocol ref: 19/SC/0217 2.0. Accessed June 2022.https://www.southampton.ac.uk/~assets/doc/brainuk/Form%20Store/BRAIN%20UK%20Protocol.pdf

4. BRAIN UK: Accessing NHS tissue archives for neuroscience research

5. Medical Research Council.Framework on the feedback of health‐related findings in research. Accessed June 2022.https://www.ukri.org/wp-content/uploads/2021/08/MRC-0208212-Framework-on-the-feedback-of-health-related-findings-in-research-2014.pdf

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1. Editorial;Neuropathology and Applied Neurobiology;2024-07-15

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