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Altered ultrasonic vocalization in neonatal SAPAP3-deficient mice

Tesdahl, Natalya S.a,b; King, Danielle K.a; McDaniel, Latisha N.a; Pieper, Andrew A.a,b,c,d,e,f

doi: 10.1097/WNR.0000000000000863
Integrative Systems

Ultrasonic vocalizations (USVs) in neonatal mice provide a means of modeling communication deficits in neurodevelopmental disorders. Mature mice deficient in SAP90/PSD95-associated protein 3 (SAPAP3) display compulsive grooming and anxiety-like behavior, conditions that are often associated with neurodevelopmental disorders. To date, however, aspects of neurodevelopment have not been investigated in SAPAP3-deficient mice. Here, we examined whether neonatal SAPAP3-deficient mice display altered USVs. We recorded USVs from 5-day-old sapap3 +/+ and sapap3 −/− mice, and also monitored developmental reflexes in these mice during the early postnatal period. Sapap3 −/− mice display an increase in the number and duration of USV calls relative to sapap3 +/+ littermates, despite otherwise similar developmental profiles. Thus, SAPAP3, previously well-characterized for its role in compulsive grooming, also plays a heretofore unidentified role in neonatal communication. Aberrant social communication and compulsive behavior are core symptoms of autism spectrum disorders, and these results show that SAPAP3-deficient mice may serve to model some aspects of these conditions.

Departments of aPsychiatry

bMolecular Physiology and Biophysics Graduate Program

cNeuroscience, Neurology, Free Radical and Radiation Biology, Radiation Oncology, Interdisciplinary Graduate Program in Human Toxicology

dDepartment of Veterans Affairs, University of Iowa Carver College of Medicine, Iowa City, IA

eWeill Cornell Autism Research Program, Weill Cornell Medical College

fIowa Neuroscience Institute, Pappajohn Biomedical Discovery Institute, New York, NY, USA

Correspondence to Andrew A. Pieper, MD, PhD, Department of Psychiatry, Pappajohn Biomedical Institute – PBDB 1318, University of Iowa Carver College of Medicine, 195 Newton Road, Iowa City 52246, Iowa, USA Tel: +1 319 353 5781; fax: +1 319 353 3003; e-mail:

Received May 7, 2017

Accepted June 19, 2017

© 2017 Wolters Kluwer Health | Lippincott Williams & Wilkins