Evidence map›Paper›PMID 29949065›Full record

ArticleJournal of community genetics2019

Parents' views of genetic testing and treatment of familial hypercholesterolemia in children: a qualitative study.

Karen Forrest Keenan, Robert M Finnie, William G Simpson, Lorna McKee, John Dean, Zosia Miedzybrodzka

Open access · hybridAbstract read
In one paragraph

Article in Journal of community genetics, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed
1.7field-weighted citation impact, top 17% 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

7 citing papers in PubMed, 16 citations in OpenAlex.

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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

6 authors at 3 institutions in 1 country.

Karen Forrest KeenanEpidemiology Group, University of Aberdeen, First Floor Health Sciences Building, Foresterhill, Aberdeen, AB25 2ZD, UK. k.keenan@abdn.ac.uk.ORCID http://orcid.org/0000-0003-4109-6929
Robert M FinnieDepartment of Medicine/Care of the Elderly, St. Johns Hospital, Howdon Road West, Livingston, UK.
William G SimpsonDepartment of Clinical Biochemistry, Aberdeen Royal Infirmary, Aberdeen, UK.
Lorna McKeeHealth Services Research Unit, University of Aberdeen, Third Floor Health Sciences Building, Foresterhill, Aberdeen, AB25 2ZD, UK.
John DeanMedical Genetics Group, University of Aberdeen, Polwarth Building, Aberdeen, AB25 2ZD, UK.
Zosia MiedzybrodzkaMedical Genetics Group, University of Aberdeen, Polwarth Building, Aberdeen, AB25 2ZD, UK.
University of Aberdeen · GBAberdeen Royal Infirmary · GBEdinburgh Royal Infirmary · GB

Funding

Chief Scientist Office HSRU2Chief Scientist Office PDF/10/06
6 · The paper itself

Abstract

Familial hypercholesterolemia (FH) is a serious inherited disorder, which greatly increases individuals' risk of cardiovascular disease (CVD) in adult life. However, medical treatment and lifestyle adjustments can fully restore life expectancy. Whilst European guidance advises that where there is a known family mutation genetic testing is undertaken in early childhood, the majority of the at-risk population remain untested and undiagnosed. To date, only a small number of studies have explored parents' and children's experiences of testing and treatment for FH, and little is known about interactions between health professionals, parents, and children in clinic settings. In this study, in-depth interviews were undertaken with parents who had attended a genetics and/or lipid clinic for FH with their children (n = 17). A thematic analysis revealed four main themes: undertaking early prevention, postponing treatment, parental concerns, and the importance of the wider family context. The majority of parents supported genetic testing for FH in childhood. However, although some were very supportive of following early treatment recommendations, others expressed reluctance. Importantly, some parents were concerned that inappropriate information had been shared with their children and wished that more time had been given to discuss how, when, and what to tell in advance. Future research is needed to explore the long-term outcomes for children who undertake genetic testing and early treatment for FH and to trial interventions to improve the engagement, follow-up, and support of children who are at risk, or diagnosed, with this disorder.

Indexed as

ChildrenFamilial hypercholesterolemia (FH)ParentsQualitative interviewsTreatment

Identifiers

PMID29949065
PMCPMC6325044
OpenAlexW2808691138

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.