改变诊断的健康相关研究结果的决策:来自儿科脑肿瘤的模型。
Decision making for health-related research outcomes that alter diagnosis: A model from paediatric brain tumours.
发表日期:2024 Aug
作者:
Jessica C Pickles, Kristian Aquilina, Jane Chalker, Christine Dahl, Abel Devadass, Kshitij Mankad, Ashirwad Merve, Munaza Ahmed, James A R Nicoll, Tabitha Bloom, David A Hilton, Neil J Sebire, Darren Hargrave, Thomas S Jacques
来源:
Neuropath Appl Neuro
摘要:
如何处理回顾性研究的临床可行结果的问题还没有得到很好的探讨。在神经病理学中,肿瘤分类的不断完善加剧了这个问题。我们试图为神经肿瘤学专业确定的潜在修订诊断建立一个披露阈值。作为先前研究的一部分,根据 WHO 2016 年指南对 73 个儿科脑肿瘤档案样本的诊断进行了重新分类。为了确定病理相关发现的披露阈值和临床可操作性,我们利用神经肿瘤学专家的替代临床多学科团队 (MDT) 在 BRAIN UK 的道德框架内进行了结果评估方法。MDT 确定了影响决策的关键决定因素-制定,包括患者管理的预期变化、自初次诊断以来经过的时间、患者存活的可能性以及自队列开始以来没有额外样本的情况。最终,我们的研究结果均不被认为具有临床可行性,这主要是由于该队列的历史档案和高风险性质。根据这一经验,我们制定了一个决策框架,以确定表明诊断发生变化的研究结果是否需要向相关临床团队报告。必须仔细考虑与使用档案组织进行研究以及识别可操作结果的可能性相关的道德问题经过考虑的。我们建立了一个结构化框架来评估与患者诊断相关的研究数据的可操作性。虽然我们的具体研究结果最适用于预后不良的儿童脑肿瘤组的病理学,但该模型可以适应一系列疾病环境,例如,研究依赖于回顾性组织队列的其他疾病,并且研究结果可能与对患者和家庭的影响,例如其他肿瘤类型、癫痫相关病理、遗传性疾病和退行性疾病。© 2024 作者。约翰·威利出版的神经病理学和应用神经生物学
The 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.As 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.The 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.Ethical 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.© 2024 The Author(s). Neuropathology and Applied Neurobiology published by John Wiley & Sons Ltd on behalf of British Neuropathological Society.