Low-level parental somatic mosaic SNVs in exomes from a large cohort of trios with diverse suspected Mendelian conditions.

Mendelian genomics exome sequencing parental somatic mosaicism rare variants

Journal

Genetics in medicine : official journal of the American College of Medical Genetics
ISSN: 1530-0366
Titre abrégé: Genet Med
Pays: United States
ID NLM: 9815831

Informations de publication

Date de publication:
11 2020
Historique:
received: 02 03 2020
accepted: 25 06 2020
revised: 25 06 2020
pubmed: 14 7 2020
medline: 29 4 2021
entrez: 14 7 2020
Statut: ppublish

Résumé

The goal of this study was to assess the scale of low-level parental mosaicism in exome sequencing (ES) databases. We analyzed approximately 2000 family trio ES data sets from the Baylor-Hopkins Center for Mendelian Genomics (BHCMG) and Baylor Genetics (BG). Among apparent de novo single-nucleotide variants identified in the affected probands, we selected rare unique variants with variant allele fraction (VAF) between 30% and 70% in the probands and lower than 10% in one of the parents. Of 102 candidate mosaic variants validated using amplicon-based next-generation sequencing, droplet digital polymerase chain reaction, or blocker displacement amplification, 27 (26.4%) were confirmed to be low- (VAF between 1% and 10%) or very low (VAF <1%) level mosaic. Detection precision in parental samples with two or more alternate reads was 63.6% (BHCMG) and 43.6% (BG). In nine investigated individuals, we observed variability of mosaic ratios among blood, saliva, fibroblast, buccal, hair, and urine samples. Our computational pipeline enables robust discrimination between true and false positive candidate mosaic variants and efficient detection of low-level mosaicism in ES samples. We confirm that the presence of two or more alternate reads in the parental sample is a reliable predictor of low-level parental somatic mosaicism.

Identifiants

pubmed: 32655138
doi: 10.1038/s41436-020-0897-z
pii: S1098-3600(21)00784-X
pmc: PMC7606563
mid: NIHMS1615434
doi:

Types de publication

Journal Article Research Support, N.I.H., Extramural

Langues

eng

Sous-ensembles de citation

IM

Pagination

1768-1776

Subventions

Organisme : NHGRI NIH HHS
ID : K08 HG008986
Pays : United States
Organisme : NICHD NIH HHS
ID : R01 HD087292
Pays : United States
Organisme : NHGRI NIH HHS
ID : UM1 HG006542
Pays : United States

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Auteurs

Tomasz Gambin (T)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Institute of Computer Science, Warsaw University of Technology, Warsaw, Poland.
Department of Medical Genetics, Institute of Mother and Child, Warsaw, Poland.

Qian Liu (Q)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.

Justyna A Karolak (JA)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Chair and Department of Genetics and Pharmaceutical Microbiology, Poznan University of Medical Sciences, Poznan, Poland.

Christopher M Grochowski (CM)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.

Nina G Xie (NG)

Department of Bioengineering, Rice University, Houston, TX, USA.

Lucia R Wu (LR)

Department of Bioengineering, Rice University, Houston, TX, USA.

Yan Helen Yan (YH)

NuProbe USA, Inc, Houston, TX, USA.

Ye Cao (Y)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Baylor Genetics, Houston, TX, USA.
Department of Obstetrics and Gynecology, The Chinese University of Hong Kong, Hong Kong SAR, China.

Zeynep H Coban Akdemir (ZH)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.

Theresa A Wilson (TA)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.

Shalini N Jhangiani (SN)

Human Genome Sequencing Center, Baylor College of Medicine, Houston, TX, USA.

Ed Chen (E)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.

Christine M Eng (CM)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Baylor Genetics, Houston, TX, USA.

Donna Muzny (D)

Human Genome Sequencing Center, Baylor College of Medicine, Houston, TX, USA.

Jennifer E Posey (JE)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.

Yaping Yang (Y)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Baylor Genetics, Houston, TX, USA.

David Y Zhang (DY)

Department of Bioengineering, Rice University, Houston, TX, USA.

Chad Shaw (C)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Baylor Genetics, Houston, TX, USA.
Department of Statistics, Rice University, Houston, TX, USA.

Pengfei Liu (P)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Baylor Genetics, Houston, TX, USA.

James R Lupski (JR)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
Baylor Genetics, Houston, TX, USA.
Texas Children's Hospital, Houston, TX, USA.
Department of Pediatrics, Baylor College of Medicine, Houston, TX, USA.

Paweł Stankiewicz (P)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA. pawels@bcm.edu.
Baylor Genetics, Houston, TX, USA. pawels@bcm.edu.

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