Genetic variants in the KDM6B gene are associated with neurodevelopmental delays and dysmorphic features.


Journal

American journal of medical genetics. Part A
ISSN: 1552-4833
Titre abrégé: Am J Med Genet A
Pays: United States
ID NLM: 101235741

Informations de publication

Date de publication:
07 2019
Historique:
received: 03 01 2019
revised: 29 03 2019
accepted: 01 04 2019
pubmed: 28 5 2019
medline: 9 7 2020
entrez: 25 5 2019
Statut: ppublish

Résumé

Lysine-specific demethylase 6B (KDM6B) demethylates trimethylated lysine-27 on histone H3. The methylation and demethylation of histone proteins affects gene expression during development. Pathogenic alterations in histone lysine methylation and demethylation genes have been associated with multiple neurodevelopmental disorders. We have identified a number of de novo alterations in the KDM6B gene via whole exome sequencing (WES) in a cohort of 12 unrelated patients with developmental delay, intellectual disability, dysmorphic facial features, and other clinical findings. Our findings will allow for further investigation in to the role of the KDM6B gene in human neurodevelopmental disorders.

Identifiants

pubmed: 31124279
doi: 10.1002/ajmg.a.61173
doi:

Substances chimiques

Jumonji Domain-Containing Histone Demethylases EC 1.14.11.-
KDM6B protein, human EC 1.14.11.-

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1276-1286

Informations de copyright

© 2019 Wiley Periodicals, Inc.

Auteurs

Elliot S Stolerman (ES)

Greenwood Genetic Center, Greenwood, South Carolina.

Elizabeth Francisco (E)

Greenwood Genetic Center, Greenwood, South Carolina.

Jennifer L Stallworth (JL)

Greenwood Genetic Center, Greenwood, South Carolina.

Julie R Jones (JR)

Greenwood Genetic Center, Greenwood, South Carolina.

Kristin G Monaghan (KG)

GeneDx, Gaithersburg, Maryland.

Jennifer Keller-Ramey (J)

GeneDx, Gaithersburg, Maryland.

Richard Person (R)

GeneDx, Gaithersburg, Maryland.

Ingrid M Wentzensen (IM)

GeneDx, Gaithersburg, Maryland.

Kirsty McWalter (K)

GeneDx, Gaithersburg, Maryland.

Boris Keren (B)

APHP, Hôpital Armand Trousseau, Paris, France.

Benedicte Heron (B)

APHP, Hôpital Armand Trousseau, Paris, France.

Caroline Nava (C)

APHP, Hôpital Armand Trousseau, Paris, France.

Delphine Heron (D)

APHP, Hôpital Armand Trousseau, Paris, France.

Katherine Kim (K)

Division of Genetics, Birth Defects and Metabolism, Ann and Robert H. Lurie Children's Hospital, Chicago, Illinois.
Feinberg School of Medicine, Northwestern University, Chicago, Illinois.

Barbara Burton (B)

Division of Genetics, Birth Defects and Metabolism, Ann and Robert H. Lurie Children's Hospital, Chicago, Illinois.
Feinberg School of Medicine, Northwestern University, Chicago, Illinois.

Fatima Al-Musafri (F)

Pediatrics Department, Hamad Medical Corporation, Qatar.

Lauren O'Grady (L)

Massachusetts General Hospital, Boston, Massachusetts.

Inderneel Sahai (I)

Massachusetts General Hospital, Boston, Massachusetts.

Luis F Escobar (LF)

Medical Genetics and Neurodevelopmental Center, Peyton Manning Children's, Hospital at St. Vincent, Indianapolis, Indiana.

Marije Meuwissen (M)

Medical Genetics, University of Antwerp, Antwerp, Belgium.

Edwin Reyniers (E)

Medical Genetics, University of Antwerp, Antwerp, Belgium.

Frank Kooy (F)

Medical Genetics, University of Antwerp, Antwerp, Belgium.

Yves Lacassie (Y)

Children's Hospital New Orleans, New Orleans, Louisiana.
Louisiana State University Health Sciences Center, New Orleans, Louisiana.

Meral Gunay-Aygun (M)

Institute of Genetic Medicine, Johns Hopkins University, Baltimore, Maryland.

Krista Sondergaard Schatz (KS)

Institute of Genetic Medicine, Johns Hopkins University, Baltimore, Maryland.

Ron Hochstenbach (R)

Department of Clinical Genetics, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, The Netherlands.

Petra J G Zwijnenburg (PJG)

Department of Clinical Genetics, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, The Netherlands.

Quinten Waisfisz (Q)

Department of Clinical Genetics, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, The Netherlands.

Marjon van Slegtenhorst (M)

Department of Clinical Genetics, Erasmus MC University Medical Center, Rotterdam, The Netherlands.

Grazia M S Mancini (GMS)

Department of Clinical Genetics, Erasmus MC University Medical Center, Rotterdam, The Netherlands.

Raymond J Louie (RJ)

Greenwood Genetic Center, Greenwood, South Carolina.

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Classifications MeSH