BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice.
BAHD1 is a heterochomatinization factor recently described as a component of a multiprotein complex associated with histone deacetylases HDAC1/2. The physiological and patho-physiological functions of BAHD1 are not yet well characterized. Here, we examined the consequences of BAHD1 deficiency in the...
| Main Authors: | , , , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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Public Library of Science (PLoS)
2020-01-01
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| Series: | PLoS ONE |
| Online Access: | https://doi.org/10.1371/journal.pone.0232789 |
| _version_ | 1818582820240490496 |
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| author | Renaud Pourpre Laurent Naudon Hamid Meziane Goran Lakisic Luc Jouneau Hugo Varet Rachel Legendre Olivia Wendling Mohammed Selloum Caroline Proux Jean-Yves Coppée Yann Herault Hélène Bierne |
| author_facet | Renaud Pourpre Laurent Naudon Hamid Meziane Goran Lakisic Luc Jouneau Hugo Varet Rachel Legendre Olivia Wendling Mohammed Selloum Caroline Proux Jean-Yves Coppée Yann Herault Hélène Bierne |
| author_sort | Renaud Pourpre |
| collection | DOAJ |
| description | BAHD1 is a heterochomatinization factor recently described as a component of a multiprotein complex associated with histone deacetylases HDAC1/2. The physiological and patho-physiological functions of BAHD1 are not yet well characterized. Here, we examined the consequences of BAHD1 deficiency in the brains of male mice. While Bahd1 knockout mice had no detectable defects in brain anatomy, RNA sequencing profiling revealed about 2500 deregulated genes in Bahd1-/- brains compared to Bahd1+/+ brains. A majority of these genes were involved in nervous system development and function, behavior, metabolism and immunity. Exploration of the Allen Brain Atlas and Dropviz databases, assessing gene expression in the brain, revealed that expression of the Bahd1 gene was limited to a few territories and cell subtypes, particularly in the hippocampal formation, the isocortex and the olfactory regions. The effect of partial BAHD1 deficiency on behavior was then evaluated on Bahd1 heterozygous male mice, which have no lethal or metabolic phenotypes. Bahd1+/- mice showed anxiety-like behavior and reduced prepulse inhibition (PPI) of the startle response. Altogether, these results suggest that BAHD1 plays a role in chromatin-dependent gene regulation in a subset of brain cells and support recent evidence linking genetic alteration of BAHD1 to psychiatric disorders in a human patient. |
| first_indexed | 2024-12-16T07:55:27Z |
| format | Article |
| id | doaj.art-38043d87852f4edb931884a30409fbc4 |
| institution | Directory Open Access Journal |
| issn | 1932-6203 |
| language | English |
| last_indexed | 2024-12-16T07:55:27Z |
| publishDate | 2020-01-01 |
| publisher | Public Library of Science (PLoS) |
| record_format | Article |
| series | PLoS ONE |
| spelling | doaj.art-38043d87852f4edb931884a30409fbc42022-12-21T22:38:44ZengPublic Library of Science (PLoS)PLoS ONE1932-62032020-01-01155e023278910.1371/journal.pone.0232789BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice.Renaud PourpreLaurent NaudonHamid MezianeGoran LakisicLuc JouneauHugo VaretRachel LegendreOlivia WendlingMohammed SelloumCaroline ProuxJean-Yves CoppéeYann HeraultHélène BierneBAHD1 is a heterochomatinization factor recently described as a component of a multiprotein complex associated with histone deacetylases HDAC1/2. The physiological and patho-physiological functions of BAHD1 are not yet well characterized. Here, we examined the consequences of BAHD1 deficiency in the brains of male mice. While Bahd1 knockout mice had no detectable defects in brain anatomy, RNA sequencing profiling revealed about 2500 deregulated genes in Bahd1-/- brains compared to Bahd1+/+ brains. A majority of these genes were involved in nervous system development and function, behavior, metabolism and immunity. Exploration of the Allen Brain Atlas and Dropviz databases, assessing gene expression in the brain, revealed that expression of the Bahd1 gene was limited to a few territories and cell subtypes, particularly in the hippocampal formation, the isocortex and the olfactory regions. The effect of partial BAHD1 deficiency on behavior was then evaluated on Bahd1 heterozygous male mice, which have no lethal or metabolic phenotypes. Bahd1+/- mice showed anxiety-like behavior and reduced prepulse inhibition (PPI) of the startle response. Altogether, these results suggest that BAHD1 plays a role in chromatin-dependent gene regulation in a subset of brain cells and support recent evidence linking genetic alteration of BAHD1 to psychiatric disorders in a human patient.https://doi.org/10.1371/journal.pone.0232789 |
| spellingShingle | Renaud Pourpre Laurent Naudon Hamid Meziane Goran Lakisic Luc Jouneau Hugo Varet Rachel Legendre Olivia Wendling Mohammed Selloum Caroline Proux Jean-Yves Coppée Yann Herault Hélène Bierne BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice. PLoS ONE |
| title | BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice. |
| title_full | BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice. |
| title_fullStr | BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice. |
| title_full_unstemmed | BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice. |
| title_short | BAHD1 haploinsufficiency results in anxiety-like phenotypes in male mice. |
| title_sort | bahd1 haploinsufficiency results in anxiety like phenotypes in male mice |
| url | https://doi.org/10.1371/journal.pone.0232789 |
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