Effects of clonidine and methylphenidate on motor activity in Fmr1 knockout mice

Wrenn, Craige C.; Heitzer, Andrew M.; Roth, Alexandra; Nawrocki, Lauren; Valdovinos, Maria G.

doi:10.1016/j.neulet.2014.11.035

Cited by 15 publications

(10 citation statements)

References 37 publications

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“…Our results were somewhat unanticipated since a majority of prior studies in the related but different open field assay showed increased locomotor activity in Fmr1-KO mice (Liu et al 2011;Olmos-Serrano et al 2011;Pacey et al 2011;Peier et al 2000;Pietropaolo et al 2011;Qin et al 2005;Spencer et al 2005Spencer et al , 2011Thomas et al 2011;Uutela et al 2012;Yan et al 2004). Findings of unchanged (Baker et al 2010;Goebel-Goody et al 2012;Pietropaolo et al 2011;Spencer et al 2006Spencer et al , 2011Wrenn et al 2015;Zhao et al 2005) and even decreased (Fish et al 2013) open field locomotor activity in Fmr1-KO mice have also been reported. Open field performance of Fmr1-KO mice also varied with mouse background strain (Pietropaolo et al 2011;Spencer et al 2011).…”

Section: Dissociation Of Overall Movement In Novel Vs Home Cage Envisupporting

confidence: 64%

“…Findings of unchanged (Zhao et al ., 2005; Spencer et al ., 2006; Pietropaolo et al , 2011, Goebel-Goody et al ., 2012; Baker et al ., 2010; Spencer et al ., 2011; Wrenn et al ., 2015), and even decreased (Fish et al ., 2013) open field locomotor activity in Fmr1 -KO mice have also been reported. Open field performance of Fmr1 -KO mice also varied with mouse background strain (Pietropaolo et al ., 2011; Spencer et al ., 2011).…”

Section: Discussionmentioning

confidence: 99%

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Decreased home cage movement and oromotor impairments in adult Fmr1‐KO mice

Bonasera

Chaudoin

Goulding

et al. 2017

Genes Brain and Behavior

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Fragile X syndrome (FXS) is a common inherited disorder that significantly impacts family and patient day-to-day living across the entire lifespan. 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 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 demonstrate 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 to wildtypes. In addition, Fmr1-KO mice demonstrate 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.

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Section: Dissociation Of Overall Movement In Novel Vs Home Cage Envisupporting

confidence: 64%

Section: Discussionmentioning

confidence: 99%

Decreased home cage movement and oromotor impairments in adult Fmr1‐KO mice

Bonasera

Chaudoin

Goulding

et al. 2017

Genes Brain and Behavior

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“…In mice, methylphenidate is used to test the predictive validity of mouse models for ADHD, since it can rescue the hyperactivity of several of these mouse models ( de la Peña et al, 2018 ). In mouse models of autism, methylphenidate did not rescue hyperactivity in Fmr1 - / - mice ( Wrenn et al, 2015 ). In the first part of the present study, we highlighted that an injection of methylphenidate increased the activity of Shank2 +/+ mice, as expected in wild-type mice, but also increased the activity of Shank2 +/- and Shank2 -/- mice.…”

Section: Discussionmentioning

confidence: 99%

Shank2 Mutant Mice Display Hyperactivity Insensitive to Methylphenidate and Reduced Flexibility in Social Motivation, but Normal Social Recognition

Torquet

Chaumont

et al. 2018

Front. Mol. Neurosci.

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Mouse models of autism can be used to study evolutionarily conserved mechanisms underlying behavioral abnormalities in social communication and repetitive behaviors. SHANK genes code for synaptic scaffolding proteins at excitatory synapses and mutations in all SHANK genes have been associated with autism. Here, we present three behavioral aspects of the mutant mice deleted for exon 16 in Shank2. First, we treated Shank2 mutant mice with methylphenidate to rescue the hyperactivity. Our failure to do so suggests that the hyperactivity displayed by Shank2 mutant mice is not related to the one displayed by the typical mouse models of hyperactivity, and might be more closely related to manic-like behaviors. Second, by testing the effect of group housing and social isolation on social interest, we highlighted that Shank2 mutant mice lack the typical flexibility to modulate social interest, in comparison with wild-type littermates. Finally, we established a new protocol to test for social recognition in a social context. We used this protocol to show that Shank2 mutant mice were able to discriminate familiar and unknown conspecifics in free interactions. Altogether, these studies shed some light on specific aspects of the behavioral defects displayed by the Shank2 mouse model. Such information could be used to orient therapeutic strategies and to design more specific tests to characterize the complex behavior of mouse models of autism.

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“…In Ndrg2 de cient mice, their ADHD-like hyperactivity was not rescued by MPH [46]. Shank2 and Fmr1 mutant mice, both are the model of autism, display hyperactivity, but the injection of MPH increased their locomotion activity [47,48]. Relevant to the effect of MPH in Shank2 and Fmr1 de cient mice, hyperactivity in Trpm1 de cient mice may not be related to ADHD, but instead autism which is also one of the phenotypes in 15q13.3 microdeletion syndrome (Table S1) [5,49].…”

Section: Discussionmentioning

confidence: 99%

Mice with Mutations in Trpm1, a Gene within the locus of 15q13.3 Microdeletion Syndrome, Display Pronounced Hyperactivity and Decreased Anxiety-Like Behavior.

Hori

Ikuta

Takao

et al. 2020

Preprint

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15q13.3 microdeletion syndrome is a genetic disorder caused by a deletion of a region containing seven genes on chromosome 15, MTMR10, FAN1, TRPM1, MIR211, KLF13, OTUD7A, and CHRNA7, and characterized by a wide spectrum of psychiatric disorders. The contribution of each gene in this syndrome has been studied using mutant mouse models, but the phenotypes of these mice do not account for human phenotypes and the results are still controversial. The behavior of Trpm1−/− mice with relation to 15q13.3 microdeletion syndrome has not been investigated due to the visual impairment in these mice, which may confound the results of behavior tests that involve vision. We have now applied a comprehensive behavioral test battery to examine the relationship of TRPM1 and 15q13.3 microdeletion syndrome by using Trpm1 null mutant mice. Our data indicate abnormal behavior of Trpm1−/− mice which may explain some phenotypes of 15q13.3 microdeletion syndrome, including reduction of anxiety behavior, abnormality of social interaction, attenuation in fear memory, and hyperactivity, which is the most prominent phenotype of Trpm1 mutant mice. While the ON visual transduction pathway is impared in Trpm1−/− mice, we did not detect compensatory high sensitivities for other sensory modalities. Although Trpm1−/− mice share the same pathway for visual impairment with mGluR6−/− mice, hyperlocomotion activity has not been reported in mGluR6−/− mice. These data suggest that the phenotype of Trpm1−/− mice extends beyond that expected from visual impairment alone. This is the first evidence to associate TRPM1 with impairment of cognitive function similar to that found in the phenotypes of 15q13.3 microdeletion syndrome.

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Effects of clonidine and methylphenidate on motor activity in Fmr1 knockout mice

Cited by 15 publications

References 37 publications

Decreased home cage movement and oromotor impairments in adult Fmr1‐KO mice

Decreased home cage movement and oromotor impairments in adult Fmr1‐KO mice

Shank2 Mutant Mice Display Hyperactivity Insensitive to Methylphenidate and Reduced Flexibility in Social Motivation, but Normal Social Recognition

Mice with Mutations in Trpm1, a Gene within the locus of 15q13.3 Microdeletion Syndrome, Display Pronounced Hyperactivity and Decreased Anxiety-Like Behavior.

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