Bi-allelic variants in DOHH, catalyzing the last step of hypusine biosynthesis, are associated with a neurodevelopmental disorder.

DHPS DOHH EIF5A1 deoxyhypusine hydroxylase eIF5A hypusine microcephaly neurodevelopmental disorder post-translational modification translation

Journal

American journal of human genetics
ISSN: 1537-6605
Titre abrégé: Am J Hum Genet
Pays: United States
ID NLM: 0370475

Informations de publication

Date de publication:
04 08 2022
Historique:
received: 09 11 2021
accepted: 21 06 2022
pubmed: 21 7 2022
medline: 10 8 2022
entrez: 20 7 2022
Statut: ppublish

Résumé

Deoxyhypusine hydroxylase (DOHH) is the enzyme catalyzing the second step in the post-translational synthesis of hypusine [N

Identifiants

pubmed: 35858628
pii: S0002-9297(22)00263-4
doi: 10.1016/j.ajhg.2022.06.010
pmc: PMC9388783
pii:
doi:

Substances chimiques

hypusine 3874VXF092
Mixed Function Oxygenases EC 1.-
deoxyhypusine hydroxylase EC 1.14.99.29
Lysine K3Z4F929H6

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

1549-1558

Informations de copyright

Copyright © 2022 American Society of Human Genetics. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of interests The authors declare no competing interests.

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Auteurs

Alban Ziegler (A)

Département de Génétique Médicale, Centre Hospitalier Universitaire d'Angers, 49933, Angers France; Université d'Angers, MitoVasc Unit, UMR Centre National de la Recherche Scientifique 6015, INSERM 1083, 49000 Angers, France. Electronic address: alban.ziegler@chu-angers.fr.

Katharina Steindl (K)

Institute of Medical Genetics, University of Zurich, 8952 Schlieren-Zurich, Switzerland.

Ashleigh S Hanner (AS)

National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892-4340, USA.

Rajesh Kumar Kar (RK)

National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892-4340, USA.

Clément Prouteau (C)

Département de Génétique Médicale, Centre Hospitalier Universitaire d'Angers, 49933, Angers France.

Anne Boland (A)

Université Paris-Saclay, CEA, Centre National de Recherche en Génomique Humaine, 91057, Evry, France.

Jean Francois Deleuze (JF)

Université Paris-Saclay, CEA, Centre National de Recherche en Génomique Humaine, 91057, Evry, France.

Christine Coubes (C)

Département de Génétique Médicale, Hôpital Arnaud de Villeneuve, Centre Hospitalier-Universitaire de Montpellier, 34295 Montpellier, France.

Stéphane Bézieau (S)

Nantes Université, Centre Hospitalier Universitaire Nantes, Service de Génétique Médicale, 44000 Nantes, France; Nantes Université, Centre Hospitalier Universitaire Nantes, Centre National de la Recherche Scientifique, INSERM, l'institut du thorax, 44000 Nantes, France.

Sébastien Küry (S)

Nantes Université, Centre Hospitalier Universitaire Nantes, Service de Génétique Médicale, 44000 Nantes, France; Nantes Université, Centre Hospitalier Universitaire Nantes, Centre National de la Recherche Scientifique, INSERM, l'institut du thorax, 44000 Nantes, France.

Isabelle Maystadt (I)

Centre de Génétique Humaine, Institut de Pathologie et de Génétique, 6041 Gosselies, Belgique.

Morgane Le Mao (M)

Université d'Angers, MitoVasc Unit, UMR Centre National de la Recherche Scientifique 6015, INSERM 1083, 49000 Angers, France.

Guy Lenaers (G)

Université d'Angers, MitoVasc Unit, UMR Centre National de la Recherche Scientifique 6015, INSERM 1083, 49000 Angers, France; Service de Neurologie, Centre Hospitalier Universitaire d'Angers, 49933, Angers France.

Benjamin Navet (B)

Département de Génétique Médicale, Centre Hospitalier Universitaire d'Angers, 49933, Angers France.

Laurence Faivre (L)

Unité de Formation et de Recherche des Sciences de Santé, INSERM-Université de Bourgogne, UMR 1231, Genetics of Developmental Disorders, FHU-TRANSLAD, 21000, Dijon, France; Centre de Référence Anomalies du Développement et Syndromes Malformatifs, FHU-TRANSLAD, Hôpital d'Enfants, Centre Hospitalier Universitaire Dijon, 21000, Dijon, France.

Frédéric Tran Mau-Them (F)

Unité de Formation et de Recherche des Sciences de Santé, INSERM-Université de Bourgogne, UMR 1231, Genetics of Developmental Disorders, FHU-TRANSLAD, 21000, Dijon, France; Unité Fonctionnelle d'Innovation Diagnostique des Maladies Rares, FHU-TRANSLAD, Centre Hospitalier Universitaire Dijon Bourgogne, Dijon, France.

Paolo Zanoni (P)

Institute of Medical Genetics, University of Zurich, 8952 Schlieren-Zurich, Switzerland.

Wendy K Chung (WK)

Department of Pediatrics, Columbia University, New York, NY 10032, USA; Department of Medicine, Columbia University, New York, NY 10032, USA.

Anita Rauch (A)

Institute of Medical Genetics, University of Zurich, 8952 Schlieren-Zurich, Switzerland; University Children's Hospital Zurich, University of Zurich, Zurich, Switzerland.

Dominique Bonneau (D)

Département de Génétique Médicale, Centre Hospitalier Universitaire d'Angers, 49933, Angers France; Université d'Angers, MitoVasc Unit, UMR Centre National de la Recherche Scientifique 6015, INSERM 1083, 49000 Angers, France.

Myung Hee Park (MH)

National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892-4340, USA. Electronic address: mhpark@nih.gov.

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Classifications MeSH