Identification of amygdala-expressed genes associated with autism spectrum disorder. Herrero, M. J., Velmeshev, D., Hernandez-Pineda, D., Sethi, S., Sorrells, S., Banerjee, P., Sullivan, C., Gupta, A. R, Kriegstein, A. R, & Corbin, J. G Mol Autism, 11(1):39, May, 2020.
abstract   bibtex   
BACKGROUND: Studies of individuals with autism spectrum disorder (ASD) have revealed a strong multigenic basis with the identification of hundreds of ASD susceptibility genes. ASD is characterized by social deficits and a range of other phenotypes, implicating complex genetics and involvement of a variety of brain regions. However, how mutations and mis-expression of select gene sets are associated with the behavioral components of ASD remains unknown. We reasoned that for genes to be associated with ASD core behaviors they must be: (1) expressed in brain regions relevant to ASD social behaviors and (2) expressed during the ASD susceptible window of brain development. METHODS: Focusing on the amygdala, a brain region whose dysfunction has been highly implicated in the social component of ASD, we mined publicly available gene expression databases to identify ASD-susceptibility genes expressed during human and mouse amygdala development. We found that a large cohort of known ASD susceptibility genes is expressed in the developing human and mouse amygdala. We further performed analysis of single-nucleus RNA-seq (snRNA-seq) data from microdissected amygdala tissue from five ASD and five control human postmortem brains ranging in age from 4 to 20 years to elucidate cell type specificity of amygdala-expressed genes and their dysregulation in ASD. RESULTS: Our analyses revealed that of the high-ranking ASD susceptibility genes, 80 are expressed in both human and mouse amygdala during fetal to early postnatal stages of development. Our human snRNA-seq analyses revealed cohorts of genes with altered expression in the ASD amygdala postnatally, especially within excitatory neurons, with dysregulated expression of seven genes predicted from our datamining pipeline. LIMITATIONS: We were limited by the ages for which we were able to obtain human tissue; therefore, the results from our datamining pipeline approach will require validation, to the extent possible, in human tissue from earlier developmental stages. CONCLUSIONS: Our pipeline narrows down the number of amygdala-expressed genes possibly involved in the social pathophysiology of ASD. Our human single-nucleus gene expression analyses revealed that ASD is characterized by changes in gene expression in specific cell types in the early postnatal amygdala.
@ARTICLE{Herrero2020-vo,
  title    = "Identification of amygdala-expressed genes associated with autism
              spectrum disorder",
  author   = "Herrero, Maria Jesus and Velmeshev, Dmitry and Hernandez-Pineda,
              David and Sethi, Saarthak and Sorrells, Shawn and Banerjee, Payal
              and Sullivan, Catherine and Gupta, Abha R and Kriegstein, Arnold
              R and Corbin, Joshua G",
  abstract = "BACKGROUND: Studies of individuals with autism spectrum disorder
              (ASD) have revealed a strong multigenic basis with the
              identification of hundreds of ASD susceptibility genes. ASD is
              characterized by social deficits and a range of other phenotypes,
              implicating complex genetics and involvement of a variety of
              brain regions. However, how mutations and mis-expression of
              select gene sets are associated with the behavioral components of
              ASD remains unknown. We reasoned that for genes to be associated
              with ASD core behaviors they must be: (1) expressed in brain
              regions relevant to ASD social behaviors and (2) expressed during
              the ASD susceptible window of brain development. METHODS:
              Focusing on the amygdala, a brain region whose dysfunction has
              been highly implicated in the social component of ASD, we mined
              publicly available gene expression databases to identify
              ASD-susceptibility genes expressed during human and mouse
              amygdala development. We found that a large cohort of known ASD
              susceptibility genes is expressed in the developing human and
              mouse amygdala. We further performed analysis of single-nucleus
              RNA-seq (snRNA-seq) data from microdissected amygdala tissue from
              five ASD and five control human postmortem brains ranging in age
              from 4 to 20 years to elucidate cell type specificity of
              amygdala-expressed genes and their dysregulation in ASD. RESULTS:
              Our analyses revealed that of the high-ranking ASD susceptibility
              genes, 80 are expressed in both human and mouse amygdala during
              fetal to early postnatal stages of development. Our human
              snRNA-seq analyses revealed cohorts of genes with altered
              expression in the ASD amygdala postnatally, especially within
              excitatory neurons, with dysregulated expression of seven genes
              predicted from our datamining pipeline. LIMITATIONS: We were
              limited by the ages for which we were able to obtain human
              tissue; therefore, the results from our datamining pipeline
              approach will require validation, to the extent possible, in
              human tissue from earlier developmental stages. CONCLUSIONS: Our
              pipeline narrows down the number of amygdala-expressed genes
              possibly involved in the social pathophysiology of ASD. Our human
              single-nucleus gene expression analyses revealed that ASD is
              characterized by changes in gene expression in specific cell
              types in the early postnatal amygdala.",
  journal  = "Mol Autism",
  volume   =  11,
  number   =  1,
  pages    = "39",
  month    =  may,
  year     =  2020,
  keywords = "ASD genes; Amygdala; Autism spectrum disorder; Brain development;
              Single nucleus RNA sequencing",
  language = "en"
}

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