TY - JOUR
T1 - Monogenic mouse models of social dysfunction
T2 - Implications for autism
AU - Oddi, D.
AU - Crusio, W. E.
AU - D'Amato, F. R.
AU - Pietropaolo, S.
PY - 2013/8/15
Y1 - 2013/8/15
N2 - Autism is a pervasive disorder characterized by a complex symptomatology, based principally on social dysfunction. The disorder has a highly complex, largely genetic etiology, involving an impressive variety of genes, the precise contributions of which still remain to be determined. For this reason, a reductionist approach to the study of autism has been proposed, employing monogenic animal models of social dysfunction, either by targeting a candidate gene, or by mimicking a single-gene disorder characterized by autistic symptoms. In the present review, we discuss this monogenic approach by comparing examples of each strategy: the mu opioid receptor knock-out (KO) mouse line, which targets the opioid system (known to be involved in the control of social behaviors); and the Fmr1-KO mouse, a model for Fragile X syndrome (a neurodevelopmental syndrome that includes autistic symptoms). The autistic-relevant behavioral phenotypes of the mu-opioid and Fmr1-KO mouse lines are described here, summarizing previous work by our research group and others, but also providing novel experimental evidence. Relevant factors influencing the validity of the two models, such as sex differences and age at testing, are also addressed, permitting an extensive evaluation of the advantages and limits of monogenic mouse models for autism.
AB - Autism is a pervasive disorder characterized by a complex symptomatology, based principally on social dysfunction. The disorder has a highly complex, largely genetic etiology, involving an impressive variety of genes, the precise contributions of which still remain to be determined. For this reason, a reductionist approach to the study of autism has been proposed, employing monogenic animal models of social dysfunction, either by targeting a candidate gene, or by mimicking a single-gene disorder characterized by autistic symptoms. In the present review, we discuss this monogenic approach by comparing examples of each strategy: the mu opioid receptor knock-out (KO) mouse line, which targets the opioid system (known to be involved in the control of social behaviors); and the Fmr1-KO mouse, a model for Fragile X syndrome (a neurodevelopmental syndrome that includes autistic symptoms). The autistic-relevant behavioral phenotypes of the mu-opioid and Fmr1-KO mouse lines are described here, summarizing previous work by our research group and others, but also providing novel experimental evidence. Relevant factors influencing the validity of the two models, such as sex differences and age at testing, are also addressed, permitting an extensive evaluation of the advantages and limits of monogenic mouse models for autism.
KW - Fmr1
KW - Fragile X syndrome
KW - Mouse models
KW - Mu receptor
KW - Opioid system
KW - Social behaviors
KW - Ultrasonic communication
UR - http://www.scopus.com/inward/record.url?scp=84880331524&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=84880331524&partnerID=8YFLogxK
U2 - 10.1016/j.bbr.2013.01.002
DO - 10.1016/j.bbr.2013.01.002
M3 - Article
C2 - 23327738
AN - SCOPUS:84880331524
VL - 251
SP - 75
EP - 84
JO - Behavioural Brain Research
JF - Behavioural Brain Research
SN - 0166-4328
ER -