Bi-allelic PRRT2 variants may predispose to Self-limited Familial Infantile Epilepsy
(2024) In European Journal of Human Genetics 32(10). p.1338-1342- Abstract
Heterozygous PRRT2 variants are frequently implicated in Self-limited Infantile Epilepsy, whereas homozygous variants are so far linked to severe presentations including developmental and epileptic encephalopathy, movement disorders, and intellectual disability. In a study aiming to explore the genetics of epilepsy in the Sudanese population, we investigated several families including a consanguineous family with three siblings diagnosed with self-limited infantile epilepsy. We evaluated both dominant and recessive inheritance using whole exome sequencing and genomic arrays. We identified a pathogenic homozygous splice-site variant in the first intron of PRRT2 [NC_000016.10(NM_145239.3):c.-65-1G > A] that segregated with the... (More)
Heterozygous PRRT2 variants are frequently implicated in Self-limited Infantile Epilepsy, whereas homozygous variants are so far linked to severe presentations including developmental and epileptic encephalopathy, movement disorders, and intellectual disability. In a study aiming to explore the genetics of epilepsy in the Sudanese population, we investigated several families including a consanguineous family with three siblings diagnosed with self-limited infantile epilepsy. We evaluated both dominant and recessive inheritance using whole exome sequencing and genomic arrays. We identified a pathogenic homozygous splice-site variant in the first intron of PRRT2 [NC_000016.10(NM_145239.3):c.-65-1G > A] that segregated with the phenotype in this family. This work taps into the genetics of epilepsy in an underrepresented African population and suggests that the phenotypes of homozygous PRRT2 variants may include milder epilepsy presentations without movement disorders.
(Less)
- author
- publishing date
- 2024-10
- type
- Contribution to journal
- publication status
- published
- in
- European Journal of Human Genetics
- volume
- 32
- issue
- 10
- pages
- 1338 - 1342
- publisher
- Nature Publishing Group
- external identifiers
-
- pmid:38316952
- scopus:85184208904
- ISSN
- 1018-4813
- DOI
- 10.1038/s41431-024-01541-x
- language
- English
- LU publication?
- no
- additional info
- Publisher Copyright: © The Author(s), under exclusive licence to European Society of Human Genetics 2024.
- id
- 4d238b1a-7577-41d6-a2cb-db5552524823
- date added to LUP
- 2026-06-05 10:29:52
- date last changed
- 2026-06-19 11:49:09
@article{4d238b1a-7577-41d6-a2cb-db5552524823,
abstract = {{<p>Heterozygous PRRT2 variants are frequently implicated in Self-limited Infantile Epilepsy, whereas homozygous variants are so far linked to severe presentations including developmental and epileptic encephalopathy, movement disorders, and intellectual disability. In a study aiming to explore the genetics of epilepsy in the Sudanese population, we investigated several families including a consanguineous family with three siblings diagnosed with self-limited infantile epilepsy. We evaluated both dominant and recessive inheritance using whole exome sequencing and genomic arrays. We identified a pathogenic homozygous splice-site variant in the first intron of PRRT2 [NC_000016.10(NM_145239.3):c.-65-1G > A] that segregated with the phenotype in this family. This work taps into the genetics of epilepsy in an underrepresented African population and suggests that the phenotypes of homozygous PRRT2 variants may include milder epilepsy presentations without movement disorders.</p>}},
author = {{Koko, Mahmoud and Elseed, Maha A. and Mohammed, Inaam N. and Hamed, Ahlam A. and Abd Allah, Amal S.I. and Yahia, Ashraf and Siddig, Rayan A. and Altmüller, Janine and Toliat, Mohammad Reza and Elmahdi, Esra O. and Amin, Mutaz and Ahmed, Elhami A. and Eltazi, Isra Z.M. and Elmugadam, Fatima A. and Abdelgadir, Wasma A. and Eltaraifee, Esraa and Ibrahim, Mohamed O.M. and Ali, Nabila M.H. and Malik, Hiba M. and Babai, Arwa M. and Bakhit, Yousuf H. and Nürnberg, Peter and Ibrahim, Muntaser E. and Salih, Mustafa A. and Schubert, Julian and Elsayed, Liena E.O. and Lerche, Holger}},
issn = {{1018-4813}},
language = {{eng}},
number = {{10}},
pages = {{1338--1342}},
publisher = {{Nature Publishing Group}},
series = {{European Journal of Human Genetics}},
title = {{Bi-allelic PRRT2 variants may predispose to Self-limited Familial Infantile Epilepsy}},
url = {{http://dx.doi.org/10.1038/s41431-024-01541-x}},
doi = {{10.1038/s41431-024-01541-x}},
volume = {{32}},
year = {{2024}},
}
