biostudies-literaturePak TKBrockman FoundationNEI NIH HHSNational Eye InstituteNIA NIH HHSNIEHS NIH HHSAllen Initiative in Brain Health and Cognitive ImpairmentNIMH NIH HHSRoy J. Carver Charitable TrustLouis Stokes VA Medical Center resources and facilitiesNIGMS NIH HHSe1009484https://www.ebi.ac.uk/biostudies/studies/S-EPMC8061871biostudies-literatureUnknown17(4)Primary cilia are microtubule-based organelles present on most cells that regulate many physiological processes, ranging from maintaining energy homeostasis to renal function. However, the role of these structures in the regulation of behavior remains unknown. To study the role of cilia in behavior, we employ mouse models of the human ciliopathy, Bardet-Biedl Syndrome (BBS). Here, we demonstrate that BBS mice have significant impairments in context fear conditioning, a form of associative learning. Moreover, we show that postnatal deletion of BBS gene function, as well as congenital deletion, specifically in the forebrain, impairs context fear conditioning. Analyses indicated that these behavioral impairments are not the result of impaired hippocampal long-term potentiation. However, our rPLoS geneticsA mouse model of Bardet-Biedl Syndrome has impaired fear memory, which is rescued by lithium treatment.PMC8061871R01 EY011298EY017168R01 MH113325P30 ES005605P30 EY02558019PABH134580006-AHAR01 AG066707EY011298T32 GM007337R01 EY017168RO1AG066707Moreira NNCychosz CCSearby CTaugher RJStevens HGenova RZhang QWang KHsu YHuang SCCarter CSVogel TPak TKSheffield VCWemmie JAPieper AAfalseA mouse model of Bardet-Biedl Syndrome has impaired fear memory, which is rescued by lithium treatment.Primary cilia are microtubule-based organelles present on most cells that regulate many physiological processes, ranging from maintaining energy homeostasis to renal function. However, the role of these structures in the regulation of behavior remains unknown. To study the role of cilia in behavior, we employ mouse models of the human ciliopathy, Bardet-Biedl Syndrome (BBS). Here, we demonstrate that BBS mice have significant impairments in context fear conditioning, a form of associative learning. Moreover, we show that postnatal deletion of BBS gene function, as well as congenital deletion, specifically in the forebrain, impairs context fear conditioning. Analyses indicated that these behavioral impairments are not the result of impaired hippocampal long-term potentiation. However, our r2021-01-01T00:00:00Z2021 Apr2025-04-05T10:14:36.484Z2025-04-05T10:14:36.484ZS-EPMC80618713388653710.1371/journal.pgen.1009484