Epilepsy in KAT6A syndrome: Description of two individuals and revision of the literature.


Journal

European journal of medical genetics
ISSN: 1878-0849
Titre abrégé: Eur J Med Genet
Pays: Netherlands
ID NLM: 101247089

Informations de publication

Date de publication:
Jan 2022
Historique:
received: 16 04 2021
revised: 29 10 2021
accepted: 02 11 2021
pubmed: 9 11 2021
medline: 11 3 2022
entrez: 8 11 2021
Statut: ppublish

Résumé

Pathogenic variants in KAT6A, encoding a histone acetyltransferase, have been identified as a cause of a developmental disorder with a definite clinical spectrum including intellectual disability, speech delay, dysmorphic facial features, microcephaly, cardiac and gastrointestinal defects. Seizures have been described in a minority of patients without a detailed characterization. In this work we focus on epilepsy in KAT6A syndrome, reporting two affected girls with history of seizures, bearing a KAT6A de novo heterozygous variant, of which one is novel. We describe the different epilepsy phenotypes of these two patients and compare them to the other individuals in literature presenting with epilepsy.

Identifiants

pubmed: 34748993
pii: S1769-7212(21)00246-9
doi: 10.1016/j.ejmg.2021.104380
pii:
doi:

Substances chimiques

Histone Acetyltransferases EC 2.3.1.48
KAT6A protein, human EC 2.3.1.48

Types de publication

Case Reports Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

104380

Informations de copyright

Copyright © 2021 Elsevier Masson SAS. All rights reserved.

Auteurs

Serena Troisi (S)

Department of Neuroscience, Reproductive and Odontostomatological Sciences, Federico II University, Naples, Italy; Pediatric Neurology, Department of Neuroscience, Santobono-Pausilipon Children's Hospital, Naples, Italy. Electronic address: serena.troisi@gmail.com.

Silvia Maitz (S)

Clinical Pediatric Genetics Unit, Pediatrics Clinics, MBBM Foundation, S. Gerardo Hospital, Monza, Italy.

Mariasavina Severino (M)

Neuroradiology Unit, "IRCCS Istituto Giannina Gaslini", Genoa, Italy.

Alice Spano (A)

Clinical Pediatric Genetics Unit, Pediatrics Clinics, MBBM Foundation, S. Gerardo Hospital, Monza, Italy.

Gerarda Cappuccio (G)

Telethon Institute of Genetics and Medicine, Pozzuoli, Naples, Italy.

Nicola Brunetti-Pierri (N)

Department of Translational Medicine, Federico II University, Naples, Italy; Telethon Institute of Genetics and Medicine, Pozzuoli, Naples, Italy.

Annalaura Torella (A)

Telethon Institute of Genetics and Medicine, Pozzuoli, Naples, Italy; Department of Precision Medicine, University of Campania "Luigi Vanvitelli", Naples, Italy.

Vincenzo Nigro (V)

Telethon Institute of Genetics and Medicine, Pozzuoli, Naples, Italy; Department of Precision Medicine, University of Campania "Luigi Vanvitelli", Naples, Italy.
Telethon Undiagnosed Disease Program, Italy.

Leonilda Bilo (L)

Department of Neuroscience, Reproductive and Odontostomatological Sciences, Federico II University, Naples, Italy.

Antonietta Coppola (A)

Department of Neuroscience, Reproductive and Odontostomatological Sciences, Federico II University, Naples, Italy.

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Classifications MeSH