Molecular causes of sex-specific deficits in rodent models of neurodevelopmental disorders.
animal models
autism
neurodevelopmental disorders
sex bias
sex differences
Journal
Journal of neuroscience research
ISSN: 1097-4547
Titre abrégé: J Neurosci Res
Pays: United States
ID NLM: 7600111
Informations de publication
Date de publication:
01 2021
01 2021
Historique:
received:
14
08
2019
revised:
02
11
2019
accepted:
09
12
2019
pubmed:
25
12
2019
medline:
3
11
2021
entrez:
25
12
2019
Statut:
ppublish
Résumé
Neurodevelopmental disorders (NDDs) such as intellectual disability and autism spectrum disorder consistently show a male bias in prevalence, but it remains unclear why males and females are affected with different frequency. While many behavioral studies of transgenic NDD models have focused only on males, the requirement by the National Institutes of Health to consider sex as a biological variable has promoted the comparison of male and female performance in wild-type and mutant animals. Here, we review examples of rodent models of NDDs in which sex-specific deficits were identified in molecular, physiological, and/or behavioral responses, showing sex differences in susceptibility to disruption of genes mutated in NDDs. Haploinsufficiency in genes involved in mechanisms such as synaptic function (GABRB3 and NRXN1), chromatin remodeling (CHD8, EMHT1, and ADNP), and intracellular signaling (CC2D1A and ERK1) lead to more severe behavioral outcomes in males. However, in the absence of behavioral deficits, females can still present with cellular and electrophysiological changes that could be due to compensatory mechanisms or differential allocation of molecular and cellular functions in the two sexes. By contrasting these findings with mouse models where females are more severely affected (MTHFR and AMBRA1), we propose a framework to approach the study of sex-specific deficits possibly leading to sex bias in NDDs.
Identifiants
pubmed: 31872500
doi: 10.1002/jnr.24577
pmc: PMC7754327
doi:
Types de publication
Journal Article
Research Support, N.I.H., Extramural
Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
37-56Subventions
Organisme : NINDS NIH HHS
ID : R01 NS105000
Pays : United States
Informations de copyright
© 2019 The Authors. Journal of Neuroscience Research published by Wiley Periodicals, Inc.
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