Monogenic mouse models of social dysfunction: Implications for autism

D. Oddi, W.E. Crusio, F.R. D’Amato, S. Pietropaolo
Behavioural Brain Research. 2013-08-01; 251: 75-84
DOI: 10.1016/j.bbr.2013.01.002

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1. Behav Brain Res. 2013 Aug 15;251:75-84. doi: 10.1016/j.bbr.2013.01.002. Epub 2013
Jan 14.

Monogenic mouse models of social dysfunction: implications for autism.

Oddi D(1), Crusio WE, D’Amato FR, Pietropaolo S.

Author information:
(1)CNR, Cell Biology and Neurobiology Institute, Rome, Italy; IRCCS, Santa Lucia
Foundation, Rome, Italy.

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.

Copyright © 2013 Elsevier B.V. All rights reserved.

DOI: 10.1016/j.bbr.2013.01.002
PMID: 23327738 [Indexed for MEDLINE]


Auteurs Bordeaux Neurocampus