Temporal and spectral differences in the ultrasonic vocalizations of fragile X knock out mice during postnatal development

Lai, Jonathan; Sobala-Drozdowski, Monica; Zhou, Linghong Linda; Doering, Laurie C.; Faure, Paul; Foster, Jane A.

doi:10.1016/j.bbr.2013.10.049

Cited by 56 publications

(64 citation statements)

References 92 publications

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“…These distress calls are thought to model early vocalization abnormalities in ASD infants and are observed in several mouse models of ASD, including Fmr1 KO mice (Lai et al, 2014; Nakatani et al, 2009; Penagarikano et al, 2011; Scattoni et al, 2011). We found that Jakmip1 KO mice showed a significant increase in vocalizations and various call types across all time points (Figure 4F), not related to weight (Figure S7B).…”

Section: Resultsmentioning

confidence: 99%

JAKMIP1, a Novel Regulator of Neuronal Translation, Modulates Synaptic Function and Autistic-like Behaviors in Mouse

Berg

Lee

Chen

et al. 2015

Neuron

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SUMMARY Autism spectrum disorder is a heritable, common neurodevelopmental disorder with diverse genetic causes. Several studies have implicated protein synthesis as one among several of its potential convergent mechanisms. We originally identified janus kinase and microtubule-interacting protein 1 (JAKMIP1) as differentially expressed in patients with distinct syndromic forms of ASD, Fragile X Syndrome and 15q duplication syndrome. Here, we provide multiple lines of evidence that JAKMIP1 is a component of polyribosomes and an RNP translational regulatory complex that includes fragile X mental retardation protein, DEAD box helicase 5, and poly(A) binding protein, cytoplasmic 1. JAKMIP1 loss dysregulates neuronal translation during synaptic development, affecting glutamatergic NMDAR signaling, and results in social deficits, stereotyped activity, abnormal postnatal vocalizations, and other autistic-like behaviors in the mouse. These findings define an important and novel role for JAKMIP1 in neural development and further highlight pathways regulating mRNA translation during synaptogenesis in the genesis of neurodevelopmental disorders.

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Section: Resultsmentioning

confidence: 99%

JAKMIP1, a Novel Regulator of Neuronal Translation, Modulates Synaptic Function and Autistic-like Behaviors in Mouse

Berg

Lee

Chen

et al. 2015

Neuron

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“…After maternal separation, the type and duration of USV emitted by the Fmr1 KO pups (8 d old pups) were different from the control littermates with no apparent changes in the total number of calls (Roy et al 2012). Recently, Lai et al reported an increased number of USVs in younger (P7) Fmr1 KO pups (Lai et al 2014) suggesting that the FMRP function on communication is age-dependent.…”

Section: Fmr1 Ko Mice Show Impaired Social Interaction and Communicationmentioning

confidence: 96%

Learning and behavioral deficits associated with the absence of the fragile X mental retardation protein: what a fly and mouse model can teach us

2014

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The Fragile X syndrome (FXS) is the most frequent form of inherited mental disability and is considered a monogenic cause of autism spectrum disorder. FXS is caused by a triplet expansion that inhibits the expression of the FMR1 gene. The gene product, the Fragile X Mental Retardation Protein (FMRP), regulates mRNA metabolism in brain and nonneuronal cells. During brain development, FMRP controls the expression of key molecules involved in receptor signaling, cytoskeleton remodeling, protein synthesis and, ultimately, spine morphology. Symptoms associated with FXS include neurodevelopmental delay, cognitive impairment, anxiety, hyperactivity, and autistic-like behavior. Twenty years ago the first Fmr1 KO mouse to study FXS was generated, and several years later other key models including the mutant Drosophila melanogaster, dFmr1, have further helped the understanding of the cellular and molecular causes behind this complex syndrome. Here, we review to which extent these biological models are affected by the absence of FMRP, pointing out the similarities with the observed human dysfunction. Additionally, we discuss several potential treatments under study in animal models that are able to partially revert some of the FXS abnormalities.

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“…Benner suggested that early deprivation can induce competitive subordinance and impaired reversal learning with altered functions of the limbic and frontal cortices in adult C57BL/6 mice [14]. However, the effect of MD in early life, particularly the neonatal days during which multiple neural systems are established [15,16], has rarely been investigated, which may be partially due to the lack of parameters that can be quantitatively analyzed in very young rodents.…”

Section: Introductionmentioning

confidence: 99%

Maternal Deprivation Influences Pup Ultrasonic Vocalizations of C57BL/6J Mice

et al. 2016

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Maternal deprivation (MD) is frequently used as an early life stress model in rodents to investigate behavioral and neurological responses under stressful conditions. However, the effect of MD on the early postnatal development of rodents, which is when multiple neural systems become established, is rarely investigated due to methodological limitations. Ultrasonic vocalizations (USVs) are one of the few responses produced by neonatal rodents that can be quantitatively analyzed, and the quantification of USVs is regarded as a novel approach to investigate possible alterations in the neurobehavioral and emotional development of infant rodents under stress. To investigate the effect of MD on pup mice, we subjected C57BL/6J mice to MD and recorded the USVs of pups on postnatal days 1, 3, 7, 8, and 14. To determine whether the effect of MD on USVs was acute or cumulative, pre- and post-separation USV groups were included; sex differences in pup USV emission were also investigated. Our results suggest that (i) USV activity was high on postnatal days 3–8; (ii) the MD effect on USVs was acute, and a cumulative effect was not found; (iii) the MD mice vocalized more and longer than the controls at a lower frequency, and the effect was closely related to age; and (iv) female pups were more susceptible than males to the effect of MD on USV number and duration between postnatal days 3–8.

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Temporal and spectral differences in the ultrasonic vocalizations of fragile X knock out mice during postnatal development

Cited by 56 publications

References 92 publications

JAKMIP1, a Novel Regulator of Neuronal Translation, Modulates Synaptic Function and Autistic-like Behaviors in Mouse

JAKMIP1, a Novel Regulator of Neuronal Translation, Modulates Synaptic Function and Autistic-like Behaviors in Mouse

Learning and behavioral deficits associated with the absence of the fragile X mental retardation protein: what a fly and mouse model can teach us

Maternal Deprivation Influences Pup Ultrasonic Vocalizations of C57BL/6J Mice

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