Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in Mice
Autism spectrum disorder (ASD) is a neurodevelopmental disease that has intellectual disability (ID) and attention-deficit/hyperactivity disorder (ADHD) as its common comorbidities. Recent genetic and clinical studies report that KDM6B, a gene encoding a histone H3 lysine 27-specific demethylase, is...
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| Format: | Article |
| Language: | English |
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Frontiers Media S.A.
2022-05-01
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| Series: | Frontiers in Behavioral Neuroscience |
| Subjects: | |
| Online Access: | https://www.frontiersin.org/articles/10.3389/fnbeh.2022.905783/full |
| _version_ | 1817976679096647680 |
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| author | Yuen Gao Mohammad B. Aljazi Jin He |
| author_facet | Yuen Gao Mohammad B. Aljazi Jin He |
| author_sort | Yuen Gao |
| collection | DOAJ |
| description | Autism spectrum disorder (ASD) is a neurodevelopmental disease that has intellectual disability (ID) and attention-deficit/hyperactivity disorder (ADHD) as its common comorbidities. Recent genetic and clinical studies report that KDM6B, a gene encoding a histone H3 lysine 27-specific demethylase, is one of the highest ASD risk genes. However, the relationship between KDM6B mutations and neurodevelopmental diseases remains unclear. Here we use an animal model to show that genetic deletion of one Kdm6b allele in mice leads to autistic-like impaired sociability and object recognition memory. In addition, the mutant mice display markedly increased locomotor activity and impulsivity, two ADHD-like behavioral traits that are ameliorated by methylphenidate treatment. Thus, our study not only uncovers a potential causal link between disruptive KDM6B mutations and ASD/ADHD-like behavioral deficits but also provides a new mouse model for studying the cellular and molecular mechanisms underlying the Kdm6b-mutation-related neurodevelopmental diseases. |
| first_indexed | 2024-04-13T22:06:08Z |
| format | Article |
| id | doaj.art-6140da76319d4d049e7d3868170454cc |
| institution | Directory Open Access Journal |
| issn | 1662-5153 |
| language | English |
| last_indexed | 2024-04-13T22:06:08Z |
| publishDate | 2022-05-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Behavioral Neuroscience |
| spelling | doaj.art-6140da76319d4d049e7d3868170454cc2022-12-22T02:27:56ZengFrontiers Media S.A.Frontiers in Behavioral Neuroscience1662-51532022-05-011610.3389/fnbeh.2022.905783905783Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in MiceYuen GaoMohammad B. AljaziJin HeAutism spectrum disorder (ASD) is a neurodevelopmental disease that has intellectual disability (ID) and attention-deficit/hyperactivity disorder (ADHD) as its common comorbidities. Recent genetic and clinical studies report that KDM6B, a gene encoding a histone H3 lysine 27-specific demethylase, is one of the highest ASD risk genes. However, the relationship between KDM6B mutations and neurodevelopmental diseases remains unclear. Here we use an animal model to show that genetic deletion of one Kdm6b allele in mice leads to autistic-like impaired sociability and object recognition memory. In addition, the mutant mice display markedly increased locomotor activity and impulsivity, two ADHD-like behavioral traits that are ameliorated by methylphenidate treatment. Thus, our study not only uncovers a potential causal link between disruptive KDM6B mutations and ASD/ADHD-like behavioral deficits but also provides a new mouse model for studying the cellular and molecular mechanisms underlying the Kdm6b-mutation-related neurodevelopmental diseases.https://www.frontiersin.org/articles/10.3389/fnbeh.2022.905783/fullautism spectrum disorderintellectual disabilityattention deficit hyperactivity disorderKDM6Bhaploinsufficiencymouse model |
| spellingShingle | Yuen Gao Mohammad B. Aljazi Jin He Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in Mice Frontiers in Behavioral Neuroscience autism spectrum disorder intellectual disability attention deficit hyperactivity disorder KDM6B haploinsufficiency mouse model |
| title | Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in Mice |
| title_full | Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in Mice |
| title_fullStr | Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in Mice |
| title_full_unstemmed | Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in Mice |
| title_short | Kdm6b Haploinsufficiency Causes ASD/ADHD-Like Behavioral Deficits in Mice |
| title_sort | kdm6b haploinsufficiency causes asd adhd like behavioral deficits in mice |
| topic | autism spectrum disorder intellectual disability attention deficit hyperactivity disorder KDM6B haploinsufficiency mouse model |
| url | https://www.frontiersin.org/articles/10.3389/fnbeh.2022.905783/full |
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