title: Decreased home cage movement and oromotor impairments in adult Fmr1-KO mice.
abstract: Fragile X syndrome (FXS) is a common inherited disorder that significantly impacts family and patient day-to-day living across the entire life span. The childhood and adolescent behavioral consequences of FXS are well appreciated. However, there are significantly fewer studies (except those examining psychiatric comorbidities) assessing behavioral phenotypes seen in adults with FXS. Mice engineered with a genetic lesion of fragile X mental retardation 1 (Fmr1) recapitulate important molecular and neuroanatomical characteristics of FXS, and provide a means to evaluate adult behavioral phenotypes associated with FXS. We give the first description of baseline behaviors including feeding, drinking, movement and their circadian rhythms; all observed over 16 consecutive days following extensive environmental habituation in adult Fmr1-KO mutant mice. We find no genotypic changes in mouse food ingestion, feeding patterns, metabolism or circadian patterns of movement, feeding and drinking. After habituation, Fmr1-KO mice show significantly less daily movement during their active phase (the dark cycle). However, Fmr1-KO mice have more bouts of activity during the light cycle compared with wild types. In addition, Fmr1-KO mice show significantly less daily water ingestion during the circadian dark cycle, and this reduction in water intake is accompanied by a decrease in the amount of water ingested per lick. The observed water ingestion and circadian phenotypes noted in Fmr1-KO mice recapitulate known clinical aspects previously described in FXS. The finding of decreased movement in Fmr1-KO mice is novel, and suggests a dissociation between baseline and novelty-evoked activity for Fmr1-KO mice.
authors.affiliation: Division of Geriatrics, Department of Internal Medicine, University of Nebraska Medical Center, Omaha, NE.
authors.forename: S J
authors.identifier: 0000-0001-6808-2821
authors.initials: SJ
authors.lastname: Bonasera
authors.affiliation: Division of Geriatrics, Department of Internal Medicine, University of Nebraska Medical Center, Omaha, NE.
authors.forename: T R
authors.initials: TR
authors.lastname: Chaudoin
authors.affiliation: Department of Psychiatry and Behavioral Sciences, Northwestern University, Chicago, IL.
authors.forename: E H
authors.identifier: 0000-0003-3891-6578
authors.initials: EH
authors.lastname: Goulding
authors.affiliation: Department of Electrical Engineering, University of Nebraska-Lincoln, Lincoln, NE.
authors.forename: M
authors.initials: M
authors.lastname: Mittek
authors.affiliation: Department of Developmental Neuroscience, Monroe-Meyer Institute, University of Nebraska Medical Center, Omaha, NE, USA.
authors.forename: A
authors.identifier: 0000-0002-0172-9448
authors.initials: A
authors.lastname: Dunaevsky
mesh_terms: D000818:Animals; D002940:Circadian Rhythm; D004326:Drinking; D005247:Feeding Behavior; D051860:Fragile X Messenger Ribonucleoprotein 1 / Q000235:genetics*; D008124:Locomotion; D051379:Mice; D008810:Mice, Inbred C57BL; D010641:Phenotype
chemical_list: D051860:Fragile X Messenger Ribonucleoprotein 1; C497684:Fmr1 protein, mouse
keywords: Fmr1-KO mouse; behavior; fragile X syndrome; home cage; locomotion; water ingestion