Long-term home cage activity scans reveal lowered exploratory behaviour in symptomatic female Rett mice

Robinson, Lianne; Plano, Andrea; Cobb, Stuart; Riedel, Gernot

doi:10.1016/j.bbr.2013.04.041

Cited by 32 publications

(41 citation statements)

References 63 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Taken together these data point to a decreased environment-directed attention (cage exploration) as the main component of the general hypoactivity profile by RTT mice. These data also support previous findings in Het Mecp2 Stop/+ females [ 11 ]. Interestingly, however, such a reduced overall activity does not seem to have affected the motivation of the RTT mice to perform behavioural tests.…”

Section: Discussionsupporting

confidence: 93%

“…The evaluation of fine-motor skills and coordination in RTT mice was integrated in the present study by the characterization of the circadian locomotor profile and the time budgeting of general activities in the home-cage. The presence of a hypolocomotor profile was confirmed in RTT female mice [ 11 , 34 ]. Moreover we report here that such profile was accompanied by a reduced frequency of environment-directed behaviours, such as burrowing , digging, and bar holding episodes.…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Deficient Purposeful Use of Forepaws in Female Mice Modelling Rett Syndrome

et al. 2015

View full text Add to dashboard Cite

Rett syndrome (RTT) is a rare neurodevelopmental disorder, characterized by severe behavioural and physiological symptoms. Mutations in the methyl CpG binding protein 2 gene (MECP2) cause more than 95% of classic cases. Motor abnormalities represent a significant part of the spectrum of RTT symptoms. In the present study we investigated motor coordination and fine motor skill domains in MeCP2-308 female mice, a validated RTT model. This was complemented by the in vivo magnetic resonance spectroscopy (MRS) analysis of metabolic profile in behaviourally relevant brain areas. MeCP2-308 heterozygous female mice (Het, 10-12 months of age) were impaired in tasks validated for the assessment of purposeful and coordinated forepaw use (Morag test and Capellini handling task). A fine-grain analysis of spontaneous behaviour in the home-cage also revealed an abnormal handling pattern when interacting with the nesting material, reduced motivation to explore the environment, and increased time devoted to feeding in Het mice. The brain MRS evaluation highlighted decreased levels of bioenergetic metabolites in the striatal area in Het mice compared to controls. Present results confirm behavioural and brain alterations previously reported in MeCP2-308 males and identify novel endpoints on which the efficacy of innovative therapeutic strategies for RTT may be tested.

show abstract

Section: Discussionsupporting

confidence: 93%

Section: Discussionmentioning

confidence: 99%

Deficient Purposeful Use of Forepaws in Female Mice Modelling Rett Syndrome

et al. 2015

View full text Add to dashboard Cite

show abstract

“…While difficult to quantify, it also appeared that a subset of the mutant mice were free-running through the LD cycle. Decreased nocturnal activity has previously been reported in other Mecp2 models, with the Mecp2 Stop model showing similar decrease in overall and particularly nocturnal activity (Robinson et al, 2013) and the Mecp2 308/y model exhibiting decreased nocturnal activity without a corresponding decrease in overall ambulatory activity (Moretti et al, 2005). Disrupted daily activity Values are reported as mean ± S.E.M.…”

Section: Discussionmentioning

confidence: 87%

Circadian rhythm disruption in a mouse model of Rett syndrome circadian disruption in RTT

Loh

Kudo

et al. 2015

Neurobiology of Disease

View full text Add to dashboard Cite

“…Locomotor and circadian activity were assessed using the PhenoTyper (Noldus, The Netherlands; [19]) home cage observation system. Activity (distance moved) was recorded for 7 days and data extracted in 1-h or 10-min bins.…”

Section: Phenotyper Home Cage Activitymentioning

confidence: 99%

Knock-in of Mutated hTAU Causes Insulin Resistance, Inflammation and Proteostasis Disturbance in a Mouse Model of Frontotemporal Dementia

et al. 2019

View full text Add to dashboard Cite

Diabetes and obesity have been implicated as risk factors for dementia. However, metabolic mechanisms and associated signalling pathways have not been investigated in detail in frontotemporal dementia. We therefore here characterised physiological, behavioural and molecular phenotypes of 3-and 8-month-old male tau knock-in (PLB2 TAU) vs wild-type (PLB WT) mice. Homecage analysis suggested intact habituation but a dramatic reduction in exploratory activity in PLB2 TAU mice. Deficits in motor strength were also observed. At 3 months, PLB2 TAU mice displayed normal glucose handling but developed hyperglycaemia at 8 months, suggesting a progressive diabetic phenotype. Brain, liver and muscle tissue analyses confirmed tissue-specific deregulation of metabolic and homeostatic pathways. In brain, increased levels of phosphorylated tau and inflammation were detected alongside reduced ER regulatory markers, overall suggesting a downregulation in essential cellular defence pathways. We suggest that subtle neuronal expression of mutated human tau is sufficient to disturb systems metabolism and protein handling. Whether respective dysfunctions in tauopathy patients are also a consequence of tau pathology remains to be confirmed, but could offer new avenues for therapeutic interventions.

show abstract

Long-term home cage activity scans reveal lowered exploratory behaviour in symptomatic female Rett mice

Cited by 32 publications

References 63 publications

Deficient Purposeful Use of Forepaws in Female Mice Modelling Rett Syndrome

Deficient Purposeful Use of Forepaws in Female Mice Modelling Rett Syndrome

Circadian rhythm disruption in a mouse model of Rett syndrome circadian disruption in RTT

Knock-in of Mutated hTAU Causes Insulin Resistance, Inflammation and Proteostasis Disturbance in a Mouse Model of Frontotemporal Dementia

Contact Info

Product

Resources

About