Long-read technologies identify a hidden inverted duplication in a family with choroideremia.

CHM RNA hairpin structure choroideremia inverted duplication long-read sequencing optical genome mapping strucutural variation

Journal

HGG advances
ISSN: 2666-2477
Titre abrégé: HGG Adv
Pays: United States
ID NLM: 101772885

Informations de publication

Date de publication:
14 Oct 2021
Historique:
received: 22 04 2021
accepted: 01 07 2021
entrez: 20 1 2022
pubmed: 21 1 2022
medline: 21 1 2022
Statut: epublish

Résumé

The lack of molecular diagnoses in rare genetic diseases can be explained by limitations of current standard genomic technologies. Upcoming long-read techniques have complementary strengths to overcome these limitations, with a particular strength in identifying structural variants. By using optical genome mapping and long-read sequencing, we aimed to identify the pathogenic variant in a large family with X-linked choroideremia. In this family, aberrant splicing of exon 12 of the choroideremia gene

Identifiants

pubmed: 35047838
doi: 10.1016/j.xhgg.2021.100046
pii: S2666-2477(21)00027-0
pmc: PMC8756506
doi:

Types de publication

Journal Article

Langues

eng

Pagination

100046

Informations de copyright

© 2021 The Author(s).

Déclaration de conflit d'intérêts

The authors declare no competing interests.

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Auteurs

Zeinab Fadaie (Z)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Donders Institute for Brain, Cognition, and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands.

Kornelia Neveling (K)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, the Netherlands.

Tuomo Mantere (T)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences, Radboud University Medical Center, Nijmegen, the Netherlands.

Ronny Derks (R)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.

Lonneke Haer-Wigman (L)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.

Amber den Ouden (A)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.

Michael Kwint (M)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Donders Institute for Brain, Cognition, and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands.

Luke O'Gorman (L)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.

Dyon Valkenburg (D)

Donders Institute for Brain, Cognition, and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Ophthalmology, Radboud University Medical Center, Nijmegen, the Netherlands.

Carel B Hoyng (CB)

Donders Institute for Brain, Cognition, and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Ophthalmology, Radboud University Medical Center, Nijmegen, the Netherlands.

Christian Gilissen (C)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences, Radboud University Medical Center, Nijmegen, the Netherlands.

Lisenka E L M Vissers (LELM)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Donders Institute for Brain, Cognition, and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands.

Marcel Nelen (M)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.

Frans P M Cremers (FPM)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Donders Institute for Brain, Cognition, and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands.

Alexander Hoischen (A)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences, Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Internal Medicine and Radboud Center for Infectious Diseases (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.

Susanne Roosing (S)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Donders Institute for Brain, Cognition, and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands.

Classifications MeSH