Confined placental mosaicism is a diagnostic pitfall in dystrophinopathies: a clinical report.


Journal

European journal of human genetics : EJHG
ISSN: 1476-5438
Titre abrégé: Eur J Hum Genet
Pays: England
ID NLM: 9302235

Informations de publication

Date de publication:
16 Jul 2024
Historique:
received: 14 12 2023
accepted: 26 06 2024
revised: 04 06 2024
medline: 17 7 2024
pubmed: 17 7 2024
entrez: 16 7 2024
Statut: aheadofprint

Résumé

Single-gene copy number variants (CNVs) limited to placenta although rarely identified may have clinical implications. We describe a pregnant woman referred for chorionic villus sampling due to increased fetal nuchal translucency. Incident intragenic deletion of Duchenne muscular dystrophy (DMD) gene, affecting exons 56 and 57, was identified in a male fetus in ~23-30% of placental cells by chromosomal microarray and confirmed using multiplex ligation-dependent probe amplification (MLPA). Rapid aneuploidy testing showed normal results and the deletion was not detected in the mother. Subsequent analyses on amniotic cells yielded a normal DMD gene result, corroborating the confined placental nature of the mosaicism. Hence, this report emphasizes the importance of conducting amniocentesis following detection of mosaicism for single gene CNVs on chorionic villi, in order to preclude confined placental mosaicism (CPM). As far as we know, this report marks only the second documented situation of CPM involving an intragenic DMD deletion.

Identifiants

pubmed: 39014012
doi: 10.1038/s41431-024-01665-0
pii: 10.1038/s41431-024-01665-0
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© 2024. The Author(s), under exclusive licence to European Society of Human Genetics.

Références

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Auteurs

Quentin Sabbagh (Q)

Montpellier University, Centre de Référence « Anomalies du Développement et Syndromes Malformatifs », ERN-ITHACA, Department of Clinical Genetics, University Hospital of Montpellier, Montpellier, France. q-sabbagh@chu-montpellier.fr.

Marion Larrieux (M)

Montpellier University, Molecular Diagnostic Laboratory, University Hospital of Montpellier, Montpellier, France.

Anouck Schneider (A)

Montpellier University, Laboratory of Cytogenomics, University Hospital of Montpellier, Montpellier, France.

Corinne Theze (C)

Montpellier University, Molecular Diagnostic Laboratory, University Hospital of Montpellier, Montpellier, France.

Marie-Claire Vincent (MC)

Montpellier University, Molecular Diagnostic Laboratory, University Hospital of Montpellier, Montpellier, France.
Montpellier University, INSERM, CNRS, PhyMedExp, Montpellier, France.

Christine Coubes (C)

Montpellier University, Centre de Référence « Anomalies du Développement et Syndromes Malformatifs », ERN-ITHACA, Department of Clinical Genetics, University Hospital of Montpellier, Montpellier, France.

Jacques Puechberty (J)

Montpellier University, Laboratory of Cytogenomics, University Hospital of Montpellier, Montpellier, France.

Sarah Renard (S)

Department of Obstetrics and Gynecology, Perpignan Hospital, Perpignan, France.

Michel Koenig (M)

Montpellier University, Molecular Diagnostic Laboratory, University Hospital of Montpellier, Montpellier, France.
Montpellier University, INSERM, CNRS, PhyMedExp, Montpellier, France.

Franck Pellestor (F)

Montpellier University, Laboratory of Cytogenomics, University Hospital of Montpellier, Montpellier, France.

Mireille Cossée (M)

Montpellier University, Molecular Diagnostic Laboratory, University Hospital of Montpellier, Montpellier, France.
Montpellier University, INSERM, CNRS, PhyMedExp, Montpellier, France.

Vincent Gatinois (V)

Montpellier University, Laboratory of Cytogenomics, University Hospital of Montpellier, Montpellier, France.

Classifications MeSH