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Background:   Brinps 1–3 , and Astrotactins ( Astn )  1 and 2 , are members of the Membrane Attack Complex/Perforin (MACPF) superfamily that are predominantly expressed in the mammalian brain during development. Genetic variation at the human  BRINP2/ASTN1  and  BRINP1/ASTN2  loci has been implicated in neurodevelopmental disorders. We, and others, have previously shown that  Brinp1  −/−  mice exhibit behavior reminiscent of autism spectrum disorder (ASD) and attention deficit hyperactivity disorder (ADHD).   Method:  We created  Brinp2  −/−  mice and  Brinp3  −/−  mice via the Cre-mediated LoxP system to investigate the effect of gene deletion on anatomy and behavior. Additionally,  Brinp2  −/−  Brinp3  −/−  double knock-out mice were generated by interbreeding  Brinp2  −/−  and  Brinp3  −/−  mice. Genomic validation was carried out for each knock-out line, followed by histological, weight and behavioral examination.  Brinp1  −/−  Brinp2  −/−  Brinp3  −/−  triple knock-out mice were also generated by crossing  Brinp2/3  double knock-out mice with previously generated  Brinp1  −/−  mice, and examined by weight and histological analysis.   Results:   Brinp2  −/−  and  Brinp3  −/−  mice differ in their behavior:  Brinp2  −/−  mice are hyperactive, whereas  Brinp3  −/−  mice exhibit marked changes in anxiety-response on the elevated plus maze.  Brinp3  −/−  mice also show evidence of altered sociability. Both  Brinp2  −/−  and  Brinp3  −/−  mice have normal short-term memory, olfactory responses, pre-pulse inhibition, and motor learning. The double knock-out mice show behaviors of  Brinp2  −/−  and  Brinp3  −/−  mice, without evidence of new or exacerbated phenotypes.   Conclusion:   Brinp3  is important in moderation of anxiety, with potential relevance to anxiety disorders.  Brinp2  dysfunction resulting in hyperactivity may be relevant to the association of ADHD with chromosome locus 1q25.2.  Brinp2  −/−  and  Brinp3  −/−  genes do not compensate in the mammalian brain and likely have distinct molecular or cell-type specific functions.

Mice Lacking Brinp2 or Brinp3, or Both, Exhibit Behaviors Consistent with Neurodevelopmental Disorders