Deficits in skilled motor and auditory learning in a rat model of Rett syndrome / Katherine S. ADCOCK in Journal of Neurodevelopmental Disorders, 12 (2020)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 12 (2020) Titre : Deficits in skilled motor and auditory learning in a rat model of Rett syndrome Type de document : texte imprimé Auteurs : Katherine S. ADCOCK, Auteur ; Abigail E. BLOUNT, Auteur ; Robert A. MORRISON, Auteur ; Amanda ALVAREZ-DIEPPA, Auteur ; Michael P. KILGARD, Auteur ; Crystal T. ENGINEER, Auteur ; Seth A. HAYS, Auteur Langues : Anglais (eng) Mots-clés : Animals  Auditory Perception  Female  Learning  Methyl-CpG-Binding Protein 2/genetics  Mice  Mice, Transgenic  Rats  Rett Syndrome/complications/genetics  Auditory  MeCP2  Motor  Rett syndrome Index. décimale : PER Périodiques Résumé : BACKGROUND: Rett syndrome is an X-linked neurodevelopmental disorder caused by a mutation in the gene MECP2. Individuals with Rett syndrome display developmental regression at an early age, and develop a range of motor, auditory, cognitive, and social impairments. Several studies have successfully modeled some aspects of dysfunction and Rett syndrome-like phenotypes in transgenic mouse and rat models bearing mutations in the MECP2 gene. Here, we sought to extend these findings and characterize skilled learning, a more complex behavior known to be altered in Rett syndrome. METHODS: We evaluated the acquisition and performance of auditory and motor function on two complex tasks in heterozygous female Mecp2 rats. Animals were trained to perform a speech discrimination task or a skilled forelimb reaching task. RESULTS: Our results reveal that Mecp2 rats display slower acquisition and reduced performance on an auditory discrimination task than wild-type (WT) littermates. Similarly, Mecp2 rats exhibit impaired learning rates and worse performance on a skilled forelimb motor task compared to WT. CONCLUSIONS: Together, these findings illustrate novel deficits in skilled learning consistent with clinical manifestation of Rett syndrome and provide a framework for development of therapeutic strategies to improve these complex behaviors. En ligne : https://dx.doi.org/10.1186/s11689-020-09330-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 [article] Deficits in skilled motor and auditory learning in a rat model of Rett syndrome [texte imprimé] / Katherine S. ADCOCK, Auteur ; Abigail E. BLOUNT, Auteur ; Robert A. MORRISON, Auteur ; Amanda ALVAREZ-DIEPPA, Auteur ; Michael P. KILGARD, Auteur ; Crystal T. ENGINEER, Auteur ; Seth A. HAYS, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 12 (2020) Mots-clés : Animals  Auditory Perception  Female  Learning  Methyl-CpG-Binding Protein 2/genetics  Mice  Mice, Transgenic  Rats  Rett Syndrome/complications/genetics  Auditory  MeCP2  Motor  Rett syndrome Index. décimale : PER Périodiques Résumé : BACKGROUND: Rett syndrome is an X-linked neurodevelopmental disorder caused by a mutation in the gene MECP2. Individuals with Rett syndrome display developmental regression at an early age, and develop a range of motor, auditory, cognitive, and social impairments. Several studies have successfully modeled some aspects of dysfunction and Rett syndrome-like phenotypes in transgenic mouse and rat models bearing mutations in the MECP2 gene. Here, we sought to extend these findings and characterize skilled learning, a more complex behavior known to be altered in Rett syndrome. METHODS: We evaluated the acquisition and performance of auditory and motor function on two complex tasks in heterozygous female Mecp2 rats. Animals were trained to perform a speech discrimination task or a skilled forelimb reaching task. RESULTS: Our results reveal that Mecp2 rats display slower acquisition and reduced performance on an auditory discrimination task than wild-type (WT) littermates. Similarly, Mecp2 rats exhibit impaired learning rates and worse performance on a skilled forelimb motor task compared to WT. CONCLUSIONS: Together, these findings illustrate novel deficits in skilled learning consistent with clinical manifestation of Rett syndrome and provide a framework for development of therapeutic strategies to improve these complex behaviors. En ligne : https://dx.doi.org/10.1186/s11689-020-09330-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573

Shank3‐deficient rats exhibit degraded cortical responses to sound / Crystal T. ENGINEER in Autism Research, 11-1 (January 2018)  

Public ISBD [article]  inAutism Research > 11-1 (January 2018) . - p.59-68 Titre : Shank3‐deficient rats exhibit degraded cortical responses to sound Type de document : texte imprimé Auteurs : Crystal T. ENGINEER, Auteur ; C. RAHEBI KIMIYA, Auteur ; S. BORLAND MICHAEL, Auteur ; P. BUELL ELIZABETH, Auteur ; W. IM KWOK, Auteur ; Linda G. WILSON, Auteur ; Pryanka SHARMA, Auteur ; Sven VANNESTE, Auteur ; Hala HARONY-NICOLAS, Auteur ; Joseph D. BUXBAUM, Auteur ; Michael P. KILGARD, Auteur Article en page(s) : p.59-68 Langues : Anglais (eng) Mots-clés : Phelan?McDermid syndrome  22q13 deletion  autism  SHANK3?haploinsufficiency syndromes Index. décimale : PER Périodiques Résumé : Individuals with SHANK3 mutations have severely impaired receptive and expressive language abilities. While brain responses are known to be abnormal in these individuals, the auditory cortex response to sound has remained largely understudied. In this study, we document the auditory cortex response to speech and non speech sounds in the novel Shank3 deficient rat model. We predicted that the auditory cortex response to sounds would be impaired in Shank3 deficient rats. We found that auditory cortex responses were weaker in Shank3 heterozygous rats compared to wild?type rats. Additionally, Shank3 heterozygous responses had less spontaneous auditory cortex firing and were unable to respond well to rapid trains of noise bursts. The rat model of the auditory impairments in SHANK3 mutation could be used to test potential rehabilitation or drug therapies to improve the communication impairments observed in individuals with Phelan McDermid syndrome. Autism Res 2018, 11: 59 68. ? 2017 International Society for Autism Research, Wiley Periodicals, Inc. Lay Summary Individuals with SHANK3 mutations have severely impaired language abilities, yet the auditory cortex response to sound has remained largely understudied. In this study, we found that auditory cortex responses were weaker and were unable to respond well to rapid sounds in Shank3 deficient rats compared to control rats. The rat model of the auditory impairments in SHANK3 mutation could be used to test potential rehabilitation or drug therapies to improve the communication impairments observed in individuals with Phelan McDermid syndrome. En ligne : https://doi.org/10.1002/aur.1883 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=333 [article] Shank3‐deficient rats exhibit degraded cortical responses to sound [texte imprimé] / Crystal T. ENGINEER, Auteur ; C. RAHEBI KIMIYA, Auteur ; S. BORLAND MICHAEL, Auteur ; P. BUELL ELIZABETH, Auteur ; W. IM KWOK, Auteur ; Linda G. WILSON, Auteur ; Pryanka SHARMA, Auteur ; Sven VANNESTE, Auteur ; Hala HARONY-NICOLAS, Auteur ; Joseph D. BUXBAUM, Auteur ; Michael P. KILGARD, Auteur . - p.59-68. Langues : Anglais (eng) in Autism Research > 11-1 (January 2018) . - p.59-68 Mots-clés : Phelan?McDermid syndrome  22q13 deletion  autism  SHANK3?haploinsufficiency syndromes Index. décimale : PER Périodiques Résumé : Individuals with SHANK3 mutations have severely impaired receptive and expressive language abilities. While brain responses are known to be abnormal in these individuals, the auditory cortex response to sound has remained largely understudied. In this study, we document the auditory cortex response to speech and non speech sounds in the novel Shank3 deficient rat model. We predicted that the auditory cortex response to sounds would be impaired in Shank3 deficient rats. We found that auditory cortex responses were weaker in Shank3 heterozygous rats compared to wild?type rats. Additionally, Shank3 heterozygous responses had less spontaneous auditory cortex firing and were unable to respond well to rapid trains of noise bursts. The rat model of the auditory impairments in SHANK3 mutation could be used to test potential rehabilitation or drug therapies to improve the communication impairments observed in individuals with Phelan McDermid syndrome. Autism Res 2018, 11: 59 68. ? 2017 International Society for Autism Research, Wiley Periodicals, Inc. Lay Summary Individuals with SHANK3 mutations have severely impaired language abilities, yet the auditory cortex response to sound has remained largely understudied. In this study, we found that auditory cortex responses were weaker and were unable to respond well to rapid sounds in Shank3 deficient rats compared to control rats. The rat model of the auditory impairments in SHANK3 mutation could be used to test potential rehabilitation or drug therapies to improve the communication impairments observed in individuals with Phelan McDermid syndrome. En ligne : https://doi.org/10.1002/aur.1883 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=333

Vagus nerve stimulation as a potential adjuvant to behavioral therapy for autism and other neurodevelopmental disorders / Crystal T. ENGINEER in Journal of Neurodevelopmental Disorders, 9-1 (December 2017)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 9-1 (December 2017) . - p.20 Titre : Vagus nerve stimulation as a potential adjuvant to behavioral therapy for autism and other neurodevelopmental disorders Type de document : texte imprimé Auteurs : Crystal T. ENGINEER, Auteur ; Seth A. HAYS, Auteur ; Michael P. KILGARD, Auteur Article en page(s) : p.20 Langues : Anglais (eng) Mots-clés : Autism  Cortex  Cortical reorganization  Plasticity  Vagal nerve Index. décimale : PER Périodiques Résumé : BACKGROUND: Many children with autism and other neurodevelopmental disorders undergo expensive, time-consuming behavioral interventions that often yield only modest improvements. The development of adjunctive interventions that can increase the benefit of rehabilitation therapies is essential in order to improve the lives of individuals with neurodevelopmental disorders. MAIN TEXT: Vagus nerve stimulation (VNS) is an FDA approved therapy that is safe and effective in reducing seizure frequency and duration in individuals with epilepsy. Individuals with neurodevelopmental disorders often exhibit decreased vagal tone, and studies indicate that VNS can be used to overcome an insufficient vagal response. Multiple studies have also documented significant improvements in quality of life after VNS therapy in individuals with neurodevelopmental disorders. Moreover, recent findings indicate that VNS significantly enhances the benefits of rehabilitative training in animal models and patients, leading to greater recovery in a variety of neurological diseases. Here, we review these findings and provide a discussion of how VNS paired with rehabilitation may yield benefits in the context of neurodevelopmental disorders. CONCLUSIONS: VNS paired with behavioral therapy may represent a potential new approach to enhance rehabilitation that could significantly improve the outcomes of individuals with neurodevelopmental disorders. En ligne : http://dx.doi.org/10.1186/s11689-017-9203-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=350 [article] Vagus nerve stimulation as a potential adjuvant to behavioral therapy for autism and other neurodevelopmental disorders [texte imprimé] / Crystal T. ENGINEER, Auteur ; Seth A. HAYS, Auteur ; Michael P. KILGARD, Auteur . - p.20. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 9-1 (December 2017) . - p.20 Mots-clés : Autism  Cortex  Cortical reorganization  Plasticity  Vagal nerve Index. décimale : PER Périodiques Résumé : BACKGROUND: Many children with autism and other neurodevelopmental disorders undergo expensive, time-consuming behavioral interventions that often yield only modest improvements. The development of adjunctive interventions that can increase the benefit of rehabilitation therapies is essential in order to improve the lives of individuals with neurodevelopmental disorders. MAIN TEXT: Vagus nerve stimulation (VNS) is an FDA approved therapy that is safe and effective in reducing seizure frequency and duration in individuals with epilepsy. Individuals with neurodevelopmental disorders often exhibit decreased vagal tone, and studies indicate that VNS can be used to overcome an insufficient vagal response. Multiple studies have also documented significant improvements in quality of life after VNS therapy in individuals with neurodevelopmental disorders. Moreover, recent findings indicate that VNS significantly enhances the benefits of rehabilitative training in animal models and patients, leading to greater recovery in a variety of neurological diseases. Here, we review these findings and provide a discussion of how VNS paired with rehabilitation may yield benefits in the context of neurodevelopmental disorders. CONCLUSIONS: VNS paired with behavioral therapy may represent a potential new approach to enhance rehabilitation that could significantly improve the outcomes of individuals with neurodevelopmental disorders. En ligne : http://dx.doi.org/10.1186/s11689-017-9203-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=350

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