Evidence map›Paper›PMID 30206353›Full record

ArticleEuropean journal of human genetics : EJHG2019

Reverse pre-symptomatic testing for Huntington disease: double disclosure when 25% at-risk children reveal the genetic status to their parent.

Adeline Bonnard, Ariane Herson, Marcela Gargiulo, Alexandra Durr

Abstract read
In one paragraph

Article 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 5 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed, 1 pooled it
–field-weighted citation impact
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

5 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. Review
  3. Article
  4. Article
  5. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

4 authors.

Adeline BonnardAPHP, Genetic Department, Pitié-Salpêtrière University Hospital, Paris, France.
Ariane HersonAPHP, Genetic Department, Pitié-Salpêtrière University Hospital, Paris, France.
Marcela GargiuloAPHP, Genetic Department, Pitié-Salpêtrière University Hospital, Paris, France.
Alexandra DurrAPHP, Genetic Department, Pitié-Salpêtrière University Hospital, Paris, France. alexandra.durr@upmc.fr.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Predictive testing for Huntington disease (HD) in 25% at-risk individuals is testing with full knowledge, and sometimes assuming, that the parent does not want to know his status. The goal of this study was to understand: (1) the differences in the motivation between 25% and 50% at-risk individuals to be tested and (2) the consequences of "double disclosure", including parental reactions. Test requests from 25% at-risk individuals were rare (155/1611, 10%). We compared their motivation with those of 1456 50% at-risk individuals. The principal motivation to have the test for both groups was "to know" (48% versus 58%, p = 0.049), but the desire to have children was more frequent in the 25% at-risk group (32% versus 17%, p < 0.001). Sixty percent of the 25% at-risk group went through the testing procedure: 15% (n = 14) were variant positive for HD. Testees reported four adverse reactions of their parent (22%): one committed suicide and three became depressed. This result highlights the impact of "double disclosure", a bad result for the person themselves and the transmitting parent. It is the responsibility of the team to anticipate this outcome with the 25% at-risk individuals: children revealing the genetic status to their parent. They should help the testees and their family to find a satisfactory solution to help prevent adverse reactions. This includes ensuring that the candidate is well-infomed abour the testing options and consequences to her/himself but also to her/his parent. The at-risk parent should be offered to discuss the implications of their child's testing.

Indexed as

DisclosureParent-Child RelationsAdultChildGenetic Carrier ScreeningGenetic CounselingHumansHuntington DiseaseParents

Identifiers

PMID30206353
PMCPMC6303253

What OpenQuestion holds

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Registered trials

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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.