Diagnostic utility of whole-genome sequencing for nephronophthisis.

Molecular medicine Paediatric kidney disease

Journal

NPJ genomic medicine
ISSN: 2056-7944
Titre abrégé: NPJ Genom Med
Pays: England
ID NLM: 101685193

Informations de publication

Date de publication:
2020
Historique:
received: 13 04 2020
accepted: 19 08 2020
entrez: 7 10 2020
pubmed: 8 10 2020
medline: 8 10 2020
Statut: epublish

Résumé

Next-generation sequencing has revolutionized the molecular diagnosis of individuals affected by genetic kidney diseases. Indeed, rapid genetic testing in individuals with suspected inherited nephropathy has not only important implications for diagnosis and prognosis but also for genetic counseling. Nephronophthisis (NPHP) and related syndromes, a leading cause of end-stage renal failure, are autosomal recessive disorders characterized by the variable presentation and considerable locus heterogeneity with more than 90 genes described as single-gene causes. In this case report, we demonstrate the utility of whole-genome sequencing (WGS) for the molecular diagnosis of NPHP by identifying two putative disease-causing intronic mutations in the

Identifiants

pubmed: 33024573
doi: 10.1038/s41525-020-00147-8
pii: 10.1038/s41525-020-00147-8
pmc: PMC7506526
doi:

Types de publication

Case Reports

Langues

eng

Pagination

38

Informations de copyright

© The Author(s) 2020.

Déclaration de conflit d'intérêts

Competing interestsThe authors declare no competing interests.

Références

Pediatr Nephrol. 2018 Oct;33(10):1701-1712
pubmed: 29974258
Kidney Int. 2009 Apr;75(8):839-47
pubmed: 19177160
Am J Hum Genet. 2019 Jan 3;104(1):45-54
pubmed: 30609407
Cold Spring Harb Perspect Biol. 2017 Mar 1;9(3):
pubmed: 27793968
Genet Med. 2015 May;17(5):405-24
pubmed: 25741868
Nephrology (Carlton). 2018 Oct;23(10):904-911
pubmed: 29717526
Nat Genet. 2003 Aug;34(4):455-9
pubmed: 12872122
N Engl J Med. 2018 Oct 04;379(14):1353-1362
pubmed: 30281996
BMJ Open. 2019 Nov 3;9(11):e029699
pubmed: 31685495
EJIFCC. 2018 Apr 30;29(1):4-14
pubmed: 29765282
J Appl Genet. 2018 Aug;59(3):253-268
pubmed: 29680930
Kidney Int. 2016 Feb;89(2):468-475
pubmed: 26489029
N Engl J Med. 2011 Apr 21;364(16):1533-43
pubmed: 21506742
EJIFCC. 2018 Nov 07;29(3):215-220
pubmed: 30479607
Hum Genet. 2017 Sep;136(9):1093-1111
pubmed: 28497172
Eur J Hum Genet. 2020 Feb;28(2):165-173
pubmed: 31527858

Auteurs

Romain Larrue (R)

Service de Toxicologie et Génopathies, CHU Lille, F-59000 Lille, France.
Univ. Lille, CNRS, Inserm, CHU Lille, UMR9020- UMR-S 1277, F-59000 Lille, France.

Paul Chamley (P)

Service de Néphrologie, CHU Lille, F-59000 Lille, France.

Thomas Bardyn (T)

Service de Toxicologie et Génopathies, CHU Lille, F-59000 Lille, France.

Arnaud Lionet (A)

Service de Néphrologie, CHU Lille, F-59000 Lille, France.

Viviane Gnemmi (V)

Univ. Lille, CNRS, Inserm, CHU Lille, UMR9020- UMR-S 1277, F-59000 Lille, France.
Service d'Anatomo-pathologie, CHU Lille, F-59000 Lille, France.

Christelle Cauffiez (C)

Univ. Lille, CNRS, Inserm, CHU Lille, UMR9020- UMR-S 1277, F-59000 Lille, France.

François Glowacki (F)

Univ. Lille, CNRS, Inserm, CHU Lille, UMR9020- UMR-S 1277, F-59000 Lille, France.
Service de Néphrologie, CHU Lille, F-59000 Lille, France.

Nicolas Pottier (N)

Service de Toxicologie et Génopathies, CHU Lille, F-59000 Lille, France.
Univ. Lille, CNRS, Inserm, CHU Lille, UMR9020- UMR-S 1277, F-59000 Lille, France.

Franck Broly (F)

Service de Toxicologie et Génopathies, CHU Lille, F-59000 Lille, France.

Classifications MeSH