A further case of familial ring chromosome 20 mosaicism - molecular characterization of the ring and review of the literature.


Journal

European journal of medical genetics
ISSN: 1878-0849
Titre abrégé: Eur J Med Genet
Pays: Netherlands
ID NLM: 101247089

Informations de publication

Date de publication:
Nov 2019
Historique:
received: 02 04 2018
revised: 24 08 2018
accepted: 28 10 2018
pubmed: 6 11 2018
medline: 7 2 2020
entrez: 3 11 2018
Statut: ppublish

Résumé

Ring chromosome 20 syndrome is a rare chromosomal disorder characterized by childhood-onset drug-resistant epilepsy, behavioral problems and variable cognitive impairment. While most cases occur sporadically, parent-to-child transmission of ring 20 mosaicism has only been reported in a few exceptional families. We identified a further family with mother-to-child transmission of ring 20 mosaicism. Detailed characterization of the ring chromosome showed a complete ring with preserved telomere repetitive sequences. SNP genotyping excluded mosaic uniparental disomy and indicated that the chromosome was transmitted without recombination from mother to child. These results corroborate the findings of a previous study and support the hypothesis that inherited mosaicism is due to transmission of an unstable chromosome either prone to ring opening or to ring re-formation.

Identifiants

pubmed: 30385235
pii: S1769-7212(18)30256-8
doi: 10.1016/j.ejmg.2018.10.016
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

103564

Informations de copyright

Copyright © 2018 Elsevier Masson SAS. All rights reserved.

Auteurs

Iris Unterberger (I)

Department of Neurology, Innsbruck Medical University, Anichstraße 35, A-6020 Innsbruck, Austria.

Judith Dobesberger (J)

Department of Neurology, Innsbruck Medical University, Anichstraße 35, A-6020 Innsbruck, Austria; Department of Neurology, Christian Doppler Klinik, Paracelsus Medical University of Salzburg, Ignaz-Harrer-Straße 79, A-5020 Salzburg, Austria.

Harald Schober (H)

Department of Pediatrics, Landeskrankenhaus Feldkirch, Carinagasse 47, A-6807 Feldkirch, Austria.

Birgit Krabichler (B)

Division of Human Genetics, Department of Medical Genetics, Molecular and Clinical Pharmacology, Innsbruck Medical University, Peter-Mayr-Straße 1, A-6020 Innsbruck, Austria.

Claudia Lamina (C)

Division of Genetic Epidemiology, Department of Medical Genetics, Molecular and Clinical Pharmacology, Innsbruck Medical University, Schöpfstraße 41, A-6020 Innsbruck, Austria.

Ulrich Schatz (U)

Division of Human Genetics, Department of Medical Genetics, Molecular and Clinical Pharmacology, Innsbruck Medical University, Peter-Mayr-Straße 1, A-6020 Innsbruck, Austria.

Johannes Zschocke (J)

Division of Human Genetics, Department of Medical Genetics, Molecular and Clinical Pharmacology, Innsbruck Medical University, Peter-Mayr-Straße 1, A-6020 Innsbruck, Austria.

Gerhard Luef (G)

Department of Neurology, Innsbruck Medical University, Anichstraße 35, A-6020 Innsbruck, Austria.

Dieter Kotzot (D)

Division of Human Genetics, Department of Medical Genetics, Molecular and Clinical Pharmacology, Innsbruck Medical University, Peter-Mayr-Straße 1, A-6020 Innsbruck, Austria.

Christine Fauth (C)

Division of Human Genetics, Department of Medical Genetics, Molecular and Clinical Pharmacology, Innsbruck Medical University, Peter-Mayr-Straße 1, A-6020 Innsbruck, Austria. Electronic address: Christine.fauth@i-med.ac.at.

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Classifications MeSH