Evidence map›Paper›PMID 40418244›Full record

ArticleHuman genetics2025

Exploring mutation carriers' preferences regarding onset and progression of disease predictions for adult-onset genetic neurodegenerative diseases: a qualitative interview study.

Max J Rensink, M H N Schermer, A Tibben, E K Bijlsma, S T de Bot, J A Kievit, L L E Bolt

Abstract read
In one paragraph

Article in Human genetics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

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0cells of the map it votes in
2citing papers in PubMed
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1 · What the graph read from it

What it found

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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.

2 · The registry

The trial behind it

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Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Article
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors.

Max J RensinkMedical Ethics, Philosophy and History of Medicine, Erasmus University Medical Centre, Dr. Molewaterplein 40, Rotterdam, 3015 GD, The Netherlands. m.rensink@erasmusmc.nl.ORCID https://orcid.org/0000-0003-4799-8587
M H N SchermerMedical Ethics, Philosophy and History of Medicine, Erasmus University Medical Centre, Dr. Molewaterplein 40, Rotterdam, 3015 GD, The Netherlands.
A TibbenDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, 2333 ZA, The Netherlands.
E K BijlsmaDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, 2333 ZA, The Netherlands.
S T de BotDepartment of Neurology, Leiden University Medical Center, Leiden, 2333 ZA, The Netherlands.
J A KievitDepartment of Clinical Genetics, Erasmus University Medical Centre, Rotterdam, 3015 GD, The Netherlands.
L L E BoltMedical Ethics, Philosophy and History of Medicine, Erasmus University Medical Centre, Dr. Molewaterplein 40, Rotterdam, 3015 GD, The Netherlands.

Funding

Nederlandse Organisatie voor Wetenschappelijk Onderzoek NWA.1389.20.244
6 · The paper itself

Abstract

Currently, new research projects aim to develop prognostic models that more accurately predict the age of onset and progression of disease for adult-onset autosomal dominant neurodegenerative diseases that lack disease-modifying treatments. While such predictions can be important for medical research and valuable in clinical practice, the perspectives of mutation carriers on receiving onset and progression predictions have not yet been explored. In this Dutch qualitative interview study, conducted between May and August 2023, the preferences, views, and concerns of 25 asymptomatic mutation carriers of Huntington's Disease, Spinocerebellar Ataxia type 1, or Spinocerebellar Ataxia type 3 regarding onset and progression predictions were examined. Reasons for wanting to receive onset and progression predictions included life planning, preparing for the disease, informing family members, and reducing uncertainty and hypervigilance. Reasons against included concerns about negative psychological effects, the expectation of similar disease progression as family members, and a preference for receiving progression information at a later stage. Most participants were open towards disclosure of onset predictions but more hesitant regarding receiving progression information. The reasons expressed and the preferred predicted age ranges varied among age groups, and some differences in preferences were observed between the three diseases. These findings may guide the development and responsible implementation of such prognostic models and can support healthcare professionals in the counselling of mutation carriers of adult-onset genetic neurodegenerative diseases.

Indexed as

Huntington DiseaseMutationNeurodegenerative DiseasesAdultAgedAge of OnsetDisease ProgressionFemaleHeterozygoteHumansMaleMiddle AgedPatient PreferencePrognosisQualitative ResearchYoung Adult

Identifiers

PMID40418244
PMCPMC12170675

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