Refinements and considerations for trio whole-genome sequence analysis when investigating Mendelian diseases presenting in early childhood.

genomics mendelian disorders paediatrics rapid diagnostic whole genome rare disease

Journal

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

Informations de publication

Date de publication:
14 Jul 2022
Historique:
received: 25 01 2022
accepted: 19 04 2022
entrez: 19 5 2022
pubmed: 20 5 2022
medline: 20 5 2022
Statut: epublish

Résumé

To facilitate early deployment of whole-genome sequencing (WGS) for severely ill children, a standardized pipeline for WGS analysis with timely turnaround and primary care pediatric uptake is needed. We developed a bioinformatics pipeline for comprehensive gene-agnostic trio WGS analysis of children suspected of having an undiagnosed monogenic disease that included detection and interpretation of primary genetic mechanisms of disease, including SNVs/indels, CNVs/SVs, uniparental disomy (UPD), imprinted genes, short tandem repeat expansions, mobile element insertions,

Identifiants

pubmed: 35586607
doi: 10.1016/j.xhgg.2022.100113
pii: S2666-2477(22)00029-X
pmc: PMC9108978
doi:

Types de publication

Journal Article

Langues

eng

Pagination

100113

Informations de copyright

© 2022 University of Cambridge.

Déclaration de conflit d'intérêts

The authors declare no competing interests.

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Auteurs

Courtney E French (CE)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.
Boston Children's Hospital, Boston, MA 02115, USA.

Helen Dolling (H)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.
Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.
NIHR Bioresource, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.
Centre for Family Research, Department of Psychology, University of Cambridge, Cambridge CB2 3RQ, UK.

Karyn Mégy (K)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.
NIHR Bioresource, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Alba Sanchis-Juan (A)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.
NIHR Bioresource, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Ajay Kumar (A)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.

Isabelle Delon (I)

Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Matthew Wakeling (M)

Institute of Biomedical and Clinical Science, University of Exeter Medical School, University of Exeter, Exeter EX4 4PY, UK.

Lucy Mallin (L)

Royal Devon and Exeter NHS Foundation Trust, Exeter EX2 5DW, UK.

Shruti Agrawal (S)

Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Topun Austin (T)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.
Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Florence Walston (F)

Norfolk and Norwich University Hospital NHS Foundation Trust, Norwich NR4 7UY, UK.

Soo-Mi Park (SM)

Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Alasdair Parker (A)

Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Chinthika Piyasena (C)

Guy's and St Thomas' NHS Foundation Trust, London SE1 9RT, UK.

Kimberley Bradbury (K)

Guy's and St Thomas' NHS Foundation Trust, London SE1 9RT, UK.

Sian Ellard (S)

Institute of Biomedical and Clinical Science, University of Exeter Medical School, University of Exeter, Exeter EX4 4PY, UK.
Royal Devon and Exeter NHS Foundation Trust, Exeter EX2 5DW, UK.

David H Rowitch (DH)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.
Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.
NIHR Bioresource, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

F Lucy Raymond (FL)

School of Clinical Medicine, University of Cambridge, Cambridge CB2 1TN, UK.
Cambridge University Hospitals NHS Foundation Trust, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.
NIHR Bioresource, Cambridge Biomedical Campus, Cambridge CB2 0QQ, UK.

Classifications MeSH