Mapping behavioral landscapes in Down syndrome animal models.

Abstract

Down syndrome (DS), caused by trisomy of chromosome 21 (Hsa21), results in a spectrum of phenotypes including learning and memory deficits, motor dysfunction and social constrains. The regions on Hsa21 are conserved with their synteny on mouse chromosome 10, 16 and 17. To date, a wide range of mouse models has been developed to determine genotype-phenotype relationships and identity of the causative dosage-sensitive genes. However, the comparison of behavioral results is not obvious due to the lack of consistency in the genetics background, housing conditions and behavioral protocols used. There is a growing need to standardize some of the classical behavioral test, include automated behavioral phenotyping and sophisticated analysis techniques and move through ethologically inspired tests. Here we present an overview of the status of behavioral phenotyping of DS murine models and the limitations and possibilities to improve their characterization to address genotype-phenotype relationships for understanding the pathophysiology of DS.