Evidence map›Paper›PMID 36384545›Full record

ArticleBMC medical ethics2022

"I don't think people are ready to trust these algorithms at face value": trust and the use of machine learning algorithms in the diagnosis of rare disease.

Nina Hallowell, Shirlene Badger, Aurelia Sauerbrei, Christoffer Nellåker, Angeliki Kerasidou

Open access · goldAbstract read
In one paragraph

Article in BMC medical ethics, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.

0numbers the graph read from it
0cells of the map it votes in
18citing papers in PubMed
10.6field-weighted citation impact, top 1% of its field
1 · What the graph read from it

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.

2 · The registry

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.

3 · Its place in the literature

Who cites it

18 citing papers in PubMed, 56 citations in OpenAlex.

  1. Article
  2. Moral diversity and the challenge of responsibility in AI-CDSS.Philosophy, ethics, and humanities in medicine : PEHM · 2026
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  6. Exploring Parental Experiences of Childhood Ear Health Clinics and Their Acceptability of AI-Based Diagnostic Tools: A Qualitative Study.Health expectations : an international journal of public participation in health care and health policy · 2025
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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

5 authors at 3 institutions in 1 country.

Nina HallowellThe Ethox Centre and Wellcome Centre for Ethics and Humanities, Nuffield Department of Population Health, and Big Data Institute, University of Oxford, Oxford, UK.
Shirlene BadgerIlumina, Cambridge, UK.
Aurelia SauerbreiThe Ethox Centre and Wellcome Centre for Ethics and Humanities, Nuffield Department of Population Health, and Big Data Institute, University of Oxford, Oxford, UK. aurelia.sauerbrei@ethox.ox.ac.uk.
Christoffer NellåkerNuffield Department of Women's and Reproductive Health and Big Data Institute, University of Oxford, Oxford, UK.
Angeliki KerasidouThe Ethox Centre and Wellcome Centre for Ethics and Humanities, Nuffield Department of Population Health, and Big Data Institute, University of Oxford, Oxford, UK.
University of Oxford · GBWellcome Centre for Ethics and Humanities · GBIllumina (United Kingdom) · GB

Funding

Wellcome Trust
6 · The paper itself

Abstract

backgroundAs the use of AI becomes more pervasive, and computerised systems are used in clinical decision-making, the role of trust in, and the trustworthiness of, AI tools will need to be addressed. Using the case of computational phenotyping to support the diagnosis of rare disease in dysmorphology, this paper explores under what conditions we could place trust in medical AI tools, which employ machine learning.

methodsSemi-structured qualitative interviews (n = 20) with stakeholders (clinical geneticists, data scientists, bioinformaticians, industry and patient support group spokespersons) who design and/or work with computational phenotyping (CP) systems. The method of constant comparison was used to analyse the interview data.

resultsInterviewees emphasized the importance of establishing trust in the use of CP technology in identifying rare diseases. Trust was formulated in two interrelated ways in these data. First, interviewees talked about the importance of using CP tools within the context of a trust relationship; arguing that patients will need to trust clinicians who use AI tools and that clinicians will need to trust AI developers, if they are to adopt this technology. Second, they described a need to establish trust in the technology itself, or in the knowledge it provides-epistemic trust. Interviewees suggested CP tools used for the diagnosis of rare diseases might be perceived as more trustworthy if the user is able to vouchsafe for the technology's reliability and accuracy and the person using/developing them is trusted.

conclusionThis study suggests we need to take deliberate and meticulous steps to design reliable or confidence-worthy AI systems for use in healthcare. In addition, we need to devise reliable or confidence-worthy processes that would give rise to reliable systems; these could take the form of RCTs and/or systems of accountability transparency and responsibility that would signify the epistemic trustworthiness of these tools. words 294.

Indexed as

Rare DiseasesTrustAlgorithmsHumansMachine LearningReproducibility of ResultsAIComputational phenotypingDigital phenotypingElectronic phenotypingEpistemic and relational trustQualitative researchRare diseasesTrustworthiness

Identifiers

PMID36384545
PMCPMC9670402
OpenAlexW4309322867

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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 OpenQuestion graph.