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Acceptability of Expanded Newborn Screening to Parents in France With or Without Genetics in the First Line

Completed
Conditions
Parental Acceptability
Expanded Newborn Screening
Interventions
Other: Questionnaire
Other: Interview
Registration Number
NCT06111456
Lead Sponsor
Centre Hospitalier Universitaire Dijon
Brief Summary

The recent modifications of the French bioethics law, the therapeutic progress and the massive development of advanced genetic techniques (such Next-Generation Sequencing (NGS)) with a rapid decrease in costs imply to question the extension of Newborn Screening (NBS) to new actionable pathologies and the acceptable and relevant methods for its possible expansion. International studies are beginning to determine the potential place of NGS in NBS. In this perspective, the SeDeN project aims to fully assess the social acceptability of these issues by measuring the diversity and consistency of expectations of French health professionals, parents and public policy makers.

The SeDeN-p3 Study focuses on the opinions of parents. It aims to analyze the perception of parents in different situations: birth, early childhood, child screened in the framework of the national neonatal screening program, etc. The objective of this part is to study the understanding and expectations of parents in France regarding the extension of newborn screening as well as their preferences regarding its conditions (information, types of pathologies, screening methods, etc.).

Detailed Description

Not available

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
1585
Inclusion Criteria

All populations combined:

  • Be a parent or co-parent

  • Age of parent:

    • woman between 18 and 50 years
    • man between 18 and 60 years
  • Live in metropolitan France

  • Have received information about the SeDeN-p3 Study

  • Understand the purpose of the SeDeN-p3 Study

Self-administered questionnaire:

  • Be able to read and answer a self-administered questionnaire in French

    • Population 1Q:
  • Have a child less than a week old

  • Have just giver birth in 1 of the partner maternity hospitals during the survey period

    • Population 2:
  • Parent or co-parent whose youngest child is between 1 week and 3 years old

  • Be part of the panel of the selected survey-sample firm

Semi-structured interviews

  • Can converse fluently in French

  • Accept to conduct a recorded interview

    • Population 1E (sub-population of Population 1Q)
  • Have completed the entire questionnaire

    • Population 3
  • Have a child under 5 years old (inclusive) with 1 of the following diseases :

    • Phenylketonuria

    • Congenital hypothyroidism

    • Congenital adrenal hyperplasia

    • Cystic fibrosis

    • Sickle cell disease

    • hearing loss

    • MCAD deficiency

    • glutaric aciduria type -1

    • isovaleric academia

    • LCHAD deficiency

    • carnitine deficiency

    • homocystinuria

    • leukinosis

    • tyrosinemia type 1

      • Population 4
  • Have a child under 17 years old (inclusive), with 1 of following diseases:

    • Citrullinemia type I
    • Ornithine Transcarbamylase Deficiency
    • Methylmalonic acidaemia
    • Very long-chain acyl-CoA dehydrogenase deficiency
    • Carnitine palmitoyl transferase 1 deficiency
    • Carnitine palmitoyl transferase 2 deficiency
    • Glutaric acidaemia type II
    • Galactosaemia
    • Biotinidase deficiency
    • Pompe Disease
    • Mucopolysaccharidosis Type 1
    • Glucose-6-phophate dehydrogenase deficiency
    • X-linked Adrenoleukodystrophy
    • Spinal muscular atrophy linked to SMN1
    • S, beta-thalassemia
Exclusion Criteria
  • Have a newborn child die during the recruitment period
  • Not speak and/or understand French
  • Refuse to participate in the SeDeN-p3 Study
  • Be under judicial protection (tutelle, curatelle, habilitation familiale et sauvegarde de justice)

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Arm && Interventions
GroupInterventionDescription
Population 1QQuestionnaireParents or co-parents of a newborn child.
Population 1QInterviewParents or co-parents of a newborn child.
Population 4InterviewParents or co-parents whose child was diagnosed later based on clinical signs for diseases not included in the list of diseases screening in French newborn screening, but included in the list of diseases screened in newborn screening in other countries.
Population 2QuestionnaireParents or co-parents whose youngest child is 1 week to 3 years old.
Population 3InterviewParents or co-parents whose child had a suspicious newborn screening result that was confirmed at the diagnosis phase (except hearing).
Primary Outcome Measures
NameTimeMethod
Mixed matrix of parental acceptability dimensions of expanded newborn screening (Mixed-data matrix)November 2023

Mixed method design used to measure parental acceptability composed of quantitative and qualitative data

Secondary Outcome Measures
NameTimeMethod
Parental acceptability scores for expanded newborn screening (Theorical Framework of Acceptability scores in self-administered questionnaire to measure parental acceptability)November 2023
Measure of importance given to different modalities of information about newborn screening (by whom, when, how, etc.) (Multiple Choice Questions)November 2023
Parent opinion on newborn screening for Spinal Muscular Atrophy, Duchenne muscular dystrophy, BRCA-related breast and ovarian cancer predisposition syndrome and congenital long QTNovember 2023

(5-point Likert scale for agreement and thematic content analysis of the free comment areas)

Parent's views on the use of genetic testing in expanded newborn screening (Likert scales)November 2023
Typology of parental acceptability scores for expanded newborn screening (Classification)November 2023
Description of acceptability of expanded newborn screening to parents of a sick child (Thematic content analysis)April 2024

Trial Locations

Locations (4)

Hôpital Necker - Enfants Malades

🇫🇷

Paris, France

Hopital Antoine Beclere - Aphp

🇫🇷

Clamart, France

Chu Dijon Bourgogne

🇫🇷

Dijon, France

Groupe Hospitalier de La Haute-Saône

🇫🇷

Vesoul, France

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