Centre d'Information et de documentation du CRA Rhône-Alpes
CRA
Informations pratiques
-
Adresse
Centre d'information et de documentation
du CRA Rhône-Alpes
Centre Hospitalier le Vinatier
bât 211
95, Bd Pinel
69678 Bron CedexHoraires
Lundi au Vendredi
9h00-12h00 13h30-16h00Contact
Tél: +33(0)4 37 91 54 65
Mail
Fax: +33(0)4 37 91 54 37
-
Résultat de la recherche
20 recherche sur le mot-clé 'Animal models'
Affiner la recherche Générer le flux rss de la recherche
Partager le résultat de cette recherche Faire une suggestion
A comprehensive bioinformatics analysis of circRNA expression in the brain of distinct mouse models of Autism Spectrum Disorder / Guilherme Cordenonsi DA FONSECA ; Carmem GOTTFRIED in Research in Autism Spectrum Disorders, 109 (November 2023)
[article]
Titre : A comprehensive bioinformatics analysis of circRNA expression in the brain of distinct mouse models of Autism Spectrum Disorder Type de document : Texte imprimé et/ou numérique Auteurs : Guilherme Cordenonsi DA FONSECA, Auteur ; Carmem GOTTFRIED, Auteur Article en page(s) : 102261 Langues : Anglais (eng) Mots-clés : Autism spectrum disorder Animal models Circular RNA Bioinformatics NcRNAs Index. décimale : PER Périodiques Résumé : Background Autism Spectrum Disorder (ASD) is a neurodevelopmental condition estimated to affect 1% of individuals worldwide. Its etiology is still not fully understood, but several molecular alterations in ASD are related to noncoding RNAs (ncRNAs). Recently, gene expression of circular RNAs (circRNAs) has been shown to be altered in the brains of autistic individuals and two animal models of ASD. Here we use bioinformatics methods to analyze the gene expression of circRNAs in the brain of six distinct animal models of ASD and identify possible alterations shared between models and between models and humans. Method: six publicly available RNA-Seq data sets were used. CircRNAs were identified by a combination of algorithms using CirComPara2 software. Differentially expressed circRNAs (DECs) were evaluatedwith the Limma Voom method, and the functional profile was evaluated with clusterProfiler. Databases of risk genes and circRNAs altered in ASD were used for translational comparison. Results: We have identified over 130,000 unique circRNAs. DECs per se are shared by a few animal models. However, functional analysis revealed that the DECs from distinct models share a similar profile of changes, mostly related to synaptic structure and function. Regarding translational alterations, around 2% of DECs in mice are orthologous to DECs in autistic individuals. Conclusions: We provide an overview of the expression of circRNAs in mouse models of ASD. The analyses indicate that synaptic circRNAs may be central to the alterations found. We hope the broad identification of circRNAs can help researchers further explore their selected mouse models. En ligne : https://doi.org/10.1016/j.rasd.2023.102261 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=517
in Research in Autism Spectrum Disorders > 109 (November 2023) . - 102261[article] A comprehensive bioinformatics analysis of circRNA expression in the brain of distinct mouse models of Autism Spectrum Disorder [Texte imprimé et/ou numérique] / Guilherme Cordenonsi DA FONSECA, Auteur ; Carmem GOTTFRIED, Auteur . - 102261.
Langues : Anglais (eng)
in Research in Autism Spectrum Disorders > 109 (November 2023) . - 102261
Mots-clés : Autism spectrum disorder Animal models Circular RNA Bioinformatics NcRNAs Index. décimale : PER Périodiques Résumé : Background Autism Spectrum Disorder (ASD) is a neurodevelopmental condition estimated to affect 1% of individuals worldwide. Its etiology is still not fully understood, but several molecular alterations in ASD are related to noncoding RNAs (ncRNAs). Recently, gene expression of circular RNAs (circRNAs) has been shown to be altered in the brains of autistic individuals and two animal models of ASD. Here we use bioinformatics methods to analyze the gene expression of circRNAs in the brain of six distinct animal models of ASD and identify possible alterations shared between models and between models and humans. Method: six publicly available RNA-Seq data sets were used. CircRNAs were identified by a combination of algorithms using CirComPara2 software. Differentially expressed circRNAs (DECs) were evaluatedwith the Limma Voom method, and the functional profile was evaluated with clusterProfiler. Databases of risk genes and circRNAs altered in ASD were used for translational comparison. Results: We have identified over 130,000 unique circRNAs. DECs per se are shared by a few animal models. However, functional analysis revealed that the DECs from distinct models share a similar profile of changes, mostly related to synaptic structure and function. Regarding translational alterations, around 2% of DECs in mice are orthologous to DECs in autistic individuals. Conclusions: We provide an overview of the expression of circRNAs in mouse models of ASD. The analyses indicate that synaptic circRNAs may be central to the alterations found. We hope the broad identification of circRNAs can help researchers further explore their selected mouse models. En ligne : https://doi.org/10.1016/j.rasd.2023.102261 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=517 Genetic Effects on Cerebellar Structure Across Mouse Models of Autism Using a Magnetic Resonance Imaging Atlas / Patrick E. STEADMAN in Autism Research, 7-1 (February 2014)
[article]
Titre : Genetic Effects on Cerebellar Structure Across Mouse Models of Autism Using a Magnetic Resonance Imaging Atlas Type de document : Texte imprimé et/ou numérique Auteurs : Patrick E. STEADMAN, Auteur ; Jacob ELLEGOOD, Auteur ; Kamila U. SZULC, Auteur ; Daniel H. TURNBULL, Auteur ; Alexandra L. JOYNER, Auteur ; R. Mark HENKELMAN, Auteur ; Jason LERCH, Auteur Article en page(s) : p.124-137 Langues : Anglais (eng) Mots-clés : animal models neuroimaging neuroanatomy structural MRI genetics Index. décimale : PER Périodiques Résumé : Magnetic resonance imaging (MRI) of autism populations is confounded by the inherent heterogeneity in the individuals' genetics and environment, two factors difficult to control for. Imaging genetic animal models that recapitulate a mutation associated with autism quantify the impact of genetics on brain morphology and mitigate the confounding factors in human studies. Here, we used MRI to image three genetic mouse models with single mutations implicated in autism: Neuroligin-3 R451C knock-in, Methyl-CpG binding protein-2 (MECP2) 308-truncation and integrin ?3 homozygous knockout. This study identified the morphological differences specific to the cerebellum, a structure repeatedly linked to autism in human neuroimaging and postmortem studies. To accomplish a comparative analysis, a segmented cerebellum template was created and used to segment each study image. This template delineated 39 different cerebellar structures. For Neuroligin-3 R451C male mutants, the gray (effect size (ES)?=?1.94, FDR q?=?0.03) and white (ES?=?1.84, q?=?0.037) matter of crus II lobule and the gray matter of the paraflocculus (ES?=?1.45, q?=?0.045) were larger in volume. The MECP2 mutant mice had cerebellar volume changes that increased in scope depending on the genotype: hemizygous males to homozygous females. The integrin ?3 mutant mouse had a drastically smaller cerebellum than controls with 28 out of 39 cerebellar structures smaller. These imaging results are discussed in relation to repetitive behaviors, sociability, and learning in the context of autism. This work further illuminates the cerebellum's role in autism. En ligne : http://dx.doi.org/10.1002/aur.1344 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=227
in Autism Research > 7-1 (February 2014) . - p.124-137[article] Genetic Effects on Cerebellar Structure Across Mouse Models of Autism Using a Magnetic Resonance Imaging Atlas [Texte imprimé et/ou numérique] / Patrick E. STEADMAN, Auteur ; Jacob ELLEGOOD, Auteur ; Kamila U. SZULC, Auteur ; Daniel H. TURNBULL, Auteur ; Alexandra L. JOYNER, Auteur ; R. Mark HENKELMAN, Auteur ; Jason LERCH, Auteur . - p.124-137.
Langues : Anglais (eng)
in Autism Research > 7-1 (February 2014) . - p.124-137
Mots-clés : animal models neuroimaging neuroanatomy structural MRI genetics Index. décimale : PER Périodiques Résumé : Magnetic resonance imaging (MRI) of autism populations is confounded by the inherent heterogeneity in the individuals' genetics and environment, two factors difficult to control for. Imaging genetic animal models that recapitulate a mutation associated with autism quantify the impact of genetics on brain morphology and mitigate the confounding factors in human studies. Here, we used MRI to image three genetic mouse models with single mutations implicated in autism: Neuroligin-3 R451C knock-in, Methyl-CpG binding protein-2 (MECP2) 308-truncation and integrin ?3 homozygous knockout. This study identified the morphological differences specific to the cerebellum, a structure repeatedly linked to autism in human neuroimaging and postmortem studies. To accomplish a comparative analysis, a segmented cerebellum template was created and used to segment each study image. This template delineated 39 different cerebellar structures. For Neuroligin-3 R451C male mutants, the gray (effect size (ES)?=?1.94, FDR q?=?0.03) and white (ES?=?1.84, q?=?0.037) matter of crus II lobule and the gray matter of the paraflocculus (ES?=?1.45, q?=?0.045) were larger in volume. The MECP2 mutant mice had cerebellar volume changes that increased in scope depending on the genotype: hemizygous males to homozygous females. The integrin ?3 mutant mouse had a drastically smaller cerebellum than controls with 28 out of 39 cerebellar structures smaller. These imaging results are discussed in relation to repetitive behaviors, sociability, and learning in the context of autism. This work further illuminates the cerebellum's role in autism. En ligne : http://dx.doi.org/10.1002/aur.1344 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=227 Adult reversal of cognitive phenotypes in neurodevelopmental disorders / A. J. SILVA in Journal of Neurodevelopmental Disorders, 1-2 (June 2009)
[article]
Titre : Adult reversal of cognitive phenotypes in neurodevelopmental disorders Type de document : Texte imprimé et/ou numérique Auteurs : A. J. SILVA, Auteur ; D. EHNINGER, Auteur Article en page(s) : p.150-7 Langues : Anglais (eng) Mots-clés : Animal models Autism Neurodevelopmental disorders Rescue Treatment Index. décimale : PER Périodiques Résumé : Recent findings in mice suggest that it is possible to reverse certain neurodevelopmental disorders in adults. Changes in development, previously thought to be irreparable in adults, were believed to underlie the neurological and psychiatric phenotypes of a range of common mental health problems with a clear developmental component. As a consequence, most researchers have focused their efforts on understanding the molecular and cellular processes that alter development with the hope that early intervention could prevent the emergent pathology. Unexpectedly, several different animal model studies published recently, including animal models of autism, suggest that it may be possible to reverse neurodevelopmental disorders in adults: Addressing the underlying molecular and cellular deficits in adults could in several cases dramatically improve the neurocognitive phenotypes in these animal models. The findings reviewed here provide hope to millions of individuals afflicted with a wide range of neurodevelopmental disorders, including autism, since they suggest that it may be possible to treat or even cure them in adults. En ligne : http://dx.doi.org/10.1007/s11689-009-9018-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=341
in Journal of Neurodevelopmental Disorders > 1-2 (June 2009) . - p.150-7[article] Adult reversal of cognitive phenotypes in neurodevelopmental disorders [Texte imprimé et/ou numérique] / A. J. SILVA, Auteur ; D. EHNINGER, Auteur . - p.150-7.
Langues : Anglais (eng)
in Journal of Neurodevelopmental Disorders > 1-2 (June 2009) . - p.150-7
Mots-clés : Animal models Autism Neurodevelopmental disorders Rescue Treatment Index. décimale : PER Périodiques Résumé : Recent findings in mice suggest that it is possible to reverse certain neurodevelopmental disorders in adults. Changes in development, previously thought to be irreparable in adults, were believed to underlie the neurological and psychiatric phenotypes of a range of common mental health problems with a clear developmental component. As a consequence, most researchers have focused their efforts on understanding the molecular and cellular processes that alter development with the hope that early intervention could prevent the emergent pathology. Unexpectedly, several different animal model studies published recently, including animal models of autism, suggest that it may be possible to reverse neurodevelopmental disorders in adults: Addressing the underlying molecular and cellular deficits in adults could in several cases dramatically improve the neurocognitive phenotypes in these animal models. The findings reviewed here provide hope to millions of individuals afflicted with a wide range of neurodevelopmental disorders, including autism, since they suggest that it may be possible to treat or even cure them in adults. En ligne : http://dx.doi.org/10.1007/s11689-009-9018-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=341 Altered attentional processing in male and female rats in a prenatal valproic acid exposure model of autism spectrum disorder / Kumari ANSHU in Autism Research, 10-12 (December 2017)
[article]
Titre : Altered attentional processing in male and female rats in a prenatal valproic acid exposure model of autism spectrum disorder Type de document : Texte imprimé et/ou numérique Auteurs : Kumari ANSHU, Auteur ; Ajay Kumar NAIR, Auteur ; U. D. KUMARESAN, Auteur ; Bindu M. KUTTY, Auteur ; Shoba SRINATH, Auteur ; T. Rao LAXMI, Auteur Article en page(s) : p.1929-1944 Langues : Anglais (eng) Mots-clés : autism attention 5-choice serial reaction time task sex differences prenatal valproic acid animal models environmental risk factors Index. décimale : PER Périodiques Résumé : Attention is foundational to efficient perception and optimal goal driven behavior. Intact attentional processing is crucial for the development of social and communication skills. Deficits in attention are therefore likely contributors to the core pathophysiology of autism spectrum disorder (ASD). Clinical evidence in ASD is suggestive of impairments in attention and its control, but the underlying mechanisms remain elusive. We examined sustained, spatially divided attention in a prenatal valproic acid (VPA) model of ASD using the 5-choice serial reaction time task (5-CSRTT). As compared to controls, male and female VPA rats had progressively lower accuracy and higher omissions with increasing attentional demands during 5-CSRTT training, and showed further performance decrements when subjected to parametric task manipulations. It is noteworthy that although VPA exposure induced attentional deficits in both sexes, there were task parameter specific sex differences. Importantly, we did not find evidence of impulsivity or motivational deficits in VPA rats but we did find reduced social preference, as well as sensorimotor deficits that suggest pre-attentional information processing impairments. Importantly, with fixed rules, graded difficulty levels, and more time, VPA rats could be successfully trained on the attentional task. To the best of our knowledge, this is the first study examining attentional functions in a VPA model. Our work underscores the need for studying both sexes in ASD animal models and validates the use of the VPA model in the quest for mechanistic understanding of aberrant attentional functions and for evaluating suitable therapeutic targets. Autism Res 2017, 10: 1929–1944. © 2017 International Society for Autism Research, Wiley Periodicals, Inc. Lay Summary We studied rats prenatally exposed to valproic acid (VPA), an established rodent model of autism. Both male and female VPA rats had a range of attentional impairments with sex-specific characteristics. Importantly, with fixed rules, graded difficulty levels, and more time, VPA rats could be successfully trained on the attentional task. Our work validates the use of the VPA model in the quest for evaluating suitable therapeutic targets for improving attentional performance. En ligne : http://dx.doi.org/10.1002/aur.1852 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=323
in Autism Research > 10-12 (December 2017) . - p.1929-1944[article] Altered attentional processing in male and female rats in a prenatal valproic acid exposure model of autism spectrum disorder [Texte imprimé et/ou numérique] / Kumari ANSHU, Auteur ; Ajay Kumar NAIR, Auteur ; U. D. KUMARESAN, Auteur ; Bindu M. KUTTY, Auteur ; Shoba SRINATH, Auteur ; T. Rao LAXMI, Auteur . - p.1929-1944.
Langues : Anglais (eng)
in Autism Research > 10-12 (December 2017) . - p.1929-1944
Mots-clés : autism attention 5-choice serial reaction time task sex differences prenatal valproic acid animal models environmental risk factors Index. décimale : PER Périodiques Résumé : Attention is foundational to efficient perception and optimal goal driven behavior. Intact attentional processing is crucial for the development of social and communication skills. Deficits in attention are therefore likely contributors to the core pathophysiology of autism spectrum disorder (ASD). Clinical evidence in ASD is suggestive of impairments in attention and its control, but the underlying mechanisms remain elusive. We examined sustained, spatially divided attention in a prenatal valproic acid (VPA) model of ASD using the 5-choice serial reaction time task (5-CSRTT). As compared to controls, male and female VPA rats had progressively lower accuracy and higher omissions with increasing attentional demands during 5-CSRTT training, and showed further performance decrements when subjected to parametric task manipulations. It is noteworthy that although VPA exposure induced attentional deficits in both sexes, there were task parameter specific sex differences. Importantly, we did not find evidence of impulsivity or motivational deficits in VPA rats but we did find reduced social preference, as well as sensorimotor deficits that suggest pre-attentional information processing impairments. Importantly, with fixed rules, graded difficulty levels, and more time, VPA rats could be successfully trained on the attentional task. To the best of our knowledge, this is the first study examining attentional functions in a VPA model. Our work underscores the need for studying both sexes in ASD animal models and validates the use of the VPA model in the quest for mechanistic understanding of aberrant attentional functions and for evaluating suitable therapeutic targets. Autism Res 2017, 10: 1929–1944. © 2017 International Society for Autism Research, Wiley Periodicals, Inc. Lay Summary We studied rats prenatally exposed to valproic acid (VPA), an established rodent model of autism. Both male and female VPA rats had a range of attentional impairments with sex-specific characteristics. Importantly, with fixed rules, graded difficulty levels, and more time, VPA rats could be successfully trained on the attentional task. Our work validates the use of the VPA model in the quest for evaluating suitable therapeutic targets for improving attentional performance. En ligne : http://dx.doi.org/10.1002/aur.1852 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=323 Autism-Related Neuroligin-3 Mutation Alters Social Behavior and Spatial Learning / Thomas C. JARAMILLO in Autism Research, 7-2 (April 2014)
[article]
Titre : Autism-Related Neuroligin-3 Mutation Alters Social Behavior and Spatial Learning Type de document : Texte imprimé et/ou numérique Auteurs : Thomas C. JARAMILLO, Auteur ; Shunan LIU, Auteur ; Ami PETTERSEN, Auteur ; Shari G. BIRNBAUM, Auteur ; Craig M. POWELL, Auteur Article en page(s) : p.264-272 Mots-clés : animal models behavioral analysis of animal models intellectual disability neuroligin autism Index. décimale : PER Périodiques Résumé : Multiple candidate genes have been identified for autism spectrum disorders. While some of these genes reach genome-wide significance, others, such as the R451C point mutation in the synaptic cell adhesion molecule neuroligin-3, appear to be rare. Interestingly, two brothers with the same R451C point mutation in neuroligin-3 present clinically on seemingly disparate sides of the autism spectrum. These clinical findings suggest genetic background may play a role in modifying the penetrance of a particular autism-associated mutation. Animal models may contribute additional support for such mutations as functionally relevant and can provide mechanistic insights. Previously, in collaboration with the Südhof laboratory, we reported that mice with an R451C substitution in neuroligin-3 displayed social deficits and enhanced spatial learning. While some of these behavioral abnormalities have since been replicated independently in the Südhof laboratory, observations from the Crawley laboratory failed to replicate these findings in a similar neuroligin-3 mutant mouse model and suggested that genetic background may contribute to variation in observations across laboratories. Therefore, we sought to replicate our findings in the neuroligin-3 R451C point mutant knock-in mouse model (NL3R451C) in a different genetic background. We backcrossed our NL3R451C mouse line onto a 129S2/SvPasCrl genetic background and repeated a subset of our previous behavioral testing. NL3R451C mice on a 129S2/SvPasCrl displayed social deficits, enhanced spatial learning, and increased locomotor activity. These data extend our previous findings that NL3R451C mice exhibit autism-relevant behavioral abnormalities and further suggest that different genetic backgrounds can modify this behavioral phenotype through epistatic genetic interactions. En ligne : http://dx.doi.org/10.1002/aur.1362 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=230
in Autism Research > 7-2 (April 2014) . - p.264-272[article] Autism-Related Neuroligin-3 Mutation Alters Social Behavior and Spatial Learning [Texte imprimé et/ou numérique] / Thomas C. JARAMILLO, Auteur ; Shunan LIU, Auteur ; Ami PETTERSEN, Auteur ; Shari G. BIRNBAUM, Auteur ; Craig M. POWELL, Auteur . - p.264-272.
in Autism Research > 7-2 (April 2014) . - p.264-272
Mots-clés : animal models behavioral analysis of animal models intellectual disability neuroligin autism Index. décimale : PER Périodiques Résumé : Multiple candidate genes have been identified for autism spectrum disorders. While some of these genes reach genome-wide significance, others, such as the R451C point mutation in the synaptic cell adhesion molecule neuroligin-3, appear to be rare. Interestingly, two brothers with the same R451C point mutation in neuroligin-3 present clinically on seemingly disparate sides of the autism spectrum. These clinical findings suggest genetic background may play a role in modifying the penetrance of a particular autism-associated mutation. Animal models may contribute additional support for such mutations as functionally relevant and can provide mechanistic insights. Previously, in collaboration with the Südhof laboratory, we reported that mice with an R451C substitution in neuroligin-3 displayed social deficits and enhanced spatial learning. While some of these behavioral abnormalities have since been replicated independently in the Südhof laboratory, observations from the Crawley laboratory failed to replicate these findings in a similar neuroligin-3 mutant mouse model and suggested that genetic background may contribute to variation in observations across laboratories. Therefore, we sought to replicate our findings in the neuroligin-3 R451C point mutant knock-in mouse model (NL3R451C) in a different genetic background. We backcrossed our NL3R451C mouse line onto a 129S2/SvPasCrl genetic background and repeated a subset of our previous behavioral testing. NL3R451C mice on a 129S2/SvPasCrl displayed social deficits, enhanced spatial learning, and increased locomotor activity. These data extend our previous findings that NL3R451C mice exhibit autism-relevant behavioral abnormalities and further suggest that different genetic backgrounds can modify this behavioral phenotype through epistatic genetic interactions. En ligne : http://dx.doi.org/10.1002/aur.1362 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=230 Beyond the three-chamber test: toward a multimodal and objective assessment of social behavior in rodents / Renad JABARIN in Molecular Autism, 13 (2022)
PermalinkBrain abnormalities in a Neuroligin3 R451C knockin mouse model associated with autism / Jacob ELLEGOOD in Autism Research, 4-5 (October 2011)
PermalinkAge-specific autistic-like behaviors in heterozygous Fmr1-KO female mice / Manon GAUDUCHEAU in Autism Research, 10-6 (June 2017)
PermalinkBasal ganglia and autism – a translational perspective / Krishna SUBRAMANIAN in Autism Research, 10-11 (November 2017)
PermalinkComparative mapping of the 22q11.2 deletion region and the potential of simple model organisms / A. GUNA in Journal of Neurodevelopmental Disorders, 7-1 (December 2015)
Permalink