ReviewEuropean journal of human genetics : EJHG2024
Predicting age of onset and progression of disease in late-onset genetic neurodegenerative diseases: An ethics review and research agenda.
Review in European journal of human genetics : EJHG, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
2 citing papers in PubMed.
- November in EJHG: looking at genetic counsellor training in Europe, novel clinical guidelines and ancestral impact on variant interpretation.European journal of human genetics : EJHG · 2024Article
- Ethics of disclosure of onset-predictive biomarker test results for genetic frontotemporal dementia in the research context.Alzheimer's & dementia (Amsterdam, Netherlands)Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
3 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Currently, a prognostic biomarker-based model is being developed to predict the onset and disease progression of Huntington's Disease (HD) and Spinocerebellar Ataxia (SCA) types 1 and 3, both late-onset genetic neurodegenerative diseases lacking a disease-modifying treatment (DMT). The need for more accurate predictions of onset and disease progression arises in the context of clinical trials evaluating the effectiveness of potential DMTs and identifying the optimal time to initiate such a DMT. Moreover, such a prognostic model may provide mutation carriers with personal utility. The aim of this article is to anticipate the ethical issues raised by these new prognostic models and to formulate the ethical issues that need to be addressed to facilitate an ethically responsible development and implementation of such a model. Part one of this article describes the ethical issues raised by presymptomatic genetic testing for HD and evaluates whether and how these issues may also occur by predicting onset and disease progression. Part two presents the results of a critical interpretative review into the ethical issues raised by biomarker testing in other late-onset neurodegenerative diseases lacking a DMT. The review aims to identify new ethical issues related to biomarker testing for predicting the onset and disease progression of HD and SCA. Finally, based on parts one and two, part three proposes a research agenda for the near future regarding the most pressing ethical issues that need to be addressed to ensure an ethically responsible implementation of such a prognostic model in both research settings and clinical practice.
Indexed as
Identifiers
What Socratic holds
Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.