Evidence map›Paper›PMID 30890781›Full record

ReviewEuropean journal of human genetics : EJHG2019

A case-note review of continued pregnancies found to be at a high risk of Huntington's disease: considerations for clinical practice.

Felicity Wadrup, Simon Holden, Rhona MacLeod, Zosia Miedzybrodzka, Andrea H Németh, Shan Owens, Sara Pasalodos, Oliver Quarrell, Angus J Clarke, UK Huntington’s Disease Predictive Testing Consortium

Open access · bronzeAbstract readReview
In one paragraph

Review in European journal of human genetics : EJHG, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.7field-weighted citation impact, top 38% 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

1 citing paper in PubMed, 8 citations in OpenAlex.

  1. Article
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

10 authors at 7 institutions in 2 countries.

Felicity WadrupEast Anglian Medical Genetics Service, Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK.ORCID 0000-0001-7274-0540
Simon HoldenEast Anglian Medical Genetics Service, Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK.
Rhona MacLeodDivision of Evolution and Genomic Sciences, School of Biological Science, University of Manchester, Manchester, UK.
Zosia MiedzybrodzkaUniversity of Aberdeen College of Life Sciences and Medicine, Division of Applied Medicine, Aberdeen, UK.
Andrea H NémethOxford Centre for Genomic Medicine, Oxford University Hospitals NHS Foundation Trust, Oxford, UK.
Shan OwensHywel Dda UHB (Milford Haven Health Care Centre, Yorke St, Milford Haven, Pembrokeshire, Wales, UK.
Sara PasalodosGenomic Medicine Unit, Navarrabiomed, Biomedical Research Centre, Pamplona, Spain.
Oliver QuarrellDepartment of Clinical Genetics, Sheffield Children's Hospital, OPD II  Northern General Hospital, Herries Road, Sheffield, UK.ORCID 0000-0002-3818-9051
Angus J ClarkeInstitute of Medical Genetics, University Hospital of Wales, Cardiff, UK. clarkeaj@cardiff.ac.uk.ORCID 0000-0002-1200-9286
UK Huntington’s Disease Predictive Testing Consortium
Cambridge University Hospitals NHS Foundation Trust · GBUniversity Hospital of Wales · GBNavarrabiomed · ESNorthern General Hospital · GBUniversity of Aberdeen · GBUniversity of Manchester · GBUniversity of Oxford · GB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Huntington's disease (HD) is a severe neurodegenerative condition that impacts the whole family. Prenatal diagnosis by direct or exclusion testing is available for couples at risk of transmitting HD to their children. An ethical problem can arise after prenatal diagnosis for HD if a known 'high risk' pregnancy is continued to term: international guidelines emphasise that this situation should be avoided where possible, as it removes the resulting child's future right to make an informed, autonomous decision about predictive testing. The UK Huntington's Disease Predictive Testing Consortium recorded 21 pregnancies that were tested, identified as high-risk and then continued. In this qualitative study, health professionals reviewed the case notes of 15 of these pregnancies. This analysis generated guidelines for clinical practice. It is recommended that practitioners: (i) remind couples of the long-term consequences of continuing a high risk pregnancy, (ii) ensure couples understand the information provided, (iii) collaborate closely with other professionals involved in the couple's prenatal care, (iv) prepare couples for the procedural aspects of prenatal diagnosis and a possible termination of pregnancy, (v) allow time for in-depth pre-test counselling, (vi) explain the rationale for only making prenatal diagnosis available subject to conditions, whilst allowing for human ambivalence and acknowledging that these 'conditions' cannot be enforced, (vii) monitor the whole clinical process to ensure that it works 'smoothly', (viii) recommend couples do not disclose the result of the prenatal test to protect the confidentiality and autonomy of the future 'high-risk' child, and (ix) offer on-going contact and support.

Indexed as

AdolescentAdultFemaleGenetic CounselingGenetic TestingHumansHuntington DiseaseMalePregnancyPrenatal DiagnosisRisk FactorsYoung Adult

Identifiers

PMID30890781
PMCPMC6777622
OpenAlexW2923152521

What OpenQuestion holds

Textmetadata
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.