Literature DB >> 26321676

Degraded neural and behavioral processing of speech sounds in a rat model of Rett syndrome.

Crystal T Engineer1, Kimiya C Rahebi2, Michael S Borland2, Elizabeth P Buell2, Tracy M Centanni2, Melyssa K Fink2, Kwok W Im2, Linda G Wilson2, Michael P Kilgard2.   

Abstract

Individuals with Rett syndrome have greatly impaired speech and language abilities. Auditory brainstem responses to sounds are normal, but cortical responses are highly abnormal. In this study, we used the novel rat Mecp2 knockout model of Rett syndrome to document the neural and behavioral processing of speech sounds. We hypothesized that both speech discrimination ability and the neural response to speech sounds would be impaired in Mecp2 rats. We expected that extensive speech training would improve speech discrimination ability and the cortical response to speech sounds. Our results reveal that speech responses across all four auditory cortex fields of Mecp2 rats were hyperexcitable, responded slower, and were less able to follow rapidly presented sounds. While Mecp2 rats could accurately perform consonant and vowel discrimination tasks in quiet, they were significantly impaired at speech sound discrimination in background noise. Extensive speech training improved discrimination ability. Training shifted cortical responses in both Mecp2 and control rats to favor the onset of speech sounds. While training increased the response to low frequency sounds in control rats, the opposite occurred in Mecp2 rats. Although neural coding and plasticity are abnormal in the rat model of Rett syndrome, extensive therapy appears to be effective. These findings may help to explain some aspects of communication deficits in Rett syndrome and suggest that extensive rehabilitation therapy might prove beneficial.
Copyright © 2015 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Auditory processing; Cortex; Mecp2; Plasticity; Rett syndrome; Speech therapy

Mesh:

Substances:

Year:  2015        PMID: 26321676      PMCID: PMC4674323          DOI: 10.1016/j.nbd.2015.08.019

Source DB:  PubMed          Journal:  Neurobiol Dis        ISSN: 0969-9961            Impact factor:   5.996


  51 in total

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Authors:  Claudia A Perez; Crystal T Engineer; Vikram Jakkamsetti; Ryan S Carraway; Matthew S Perry; Michael P Kilgard
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Journal:  Neuroscience       Date:  2012-01-08       Impact factor: 3.590

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7.  Aspects of speech-language abilities are influenced by MECP2 mutation type in girls with Rett syndrome.

Authors:  Anna Urbanowicz; Jenny Downs; Sonya Girdler; Natalie Ciccone; Helen Leonard
Journal:  Am J Med Genet A       Date:  2014-11-26       Impact factor: 2.802

Review 8.  Neurophysiology of Rett syndrome.

Authors:  Daniel G Glaze
Journal:  J Child Neurol       Date:  2005-09       Impact factor: 1.987

9.  Dysfunction in GABA signalling mediates autism-like stereotypies and Rett syndrome phenotypes.

Authors:  Hsiao-Tuan Chao; Hongmei Chen; Rodney C Samaco; Mingshan Xue; Maria Chahrour; Jong Yoo; Jeffrey L Neul; Shiaoching Gong; Hui-Chen Lu; Nathaniel Heintz; Marc Ekker; John L R Rubenstein; Jeffrey L Noebels; Christian Rosenmund; Huda Y Zoghbi
Journal:  Nature       Date:  2010-11-11       Impact factor: 49.962

10.  Speech sound processing deficits and training-induced neural plasticity in rats with dyslexia gene knockdown.

Authors:  Tracy M Centanni; Fuyi Chen; Anne M Booker; Crystal T Engineer; Andrew M Sloan; Robert L Rennaker; Joseph J LoTurco; Michael P Kilgard
Journal:  PLoS One       Date:  2014-05-28       Impact factor: 3.240

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  14 in total

1.  Feasibility of event-related potential (ERP) biomarker use to study effects of mother's voice exposure on speech sound differentiation of preterm infants.

Authors:  Olena D Chorna; Ellyn L Hamm; Hemang Shrivastava; Nathalie L Maitre
Journal:  Dev Neuropsychol       Date:  2018       Impact factor: 2.253

2.  Temporal plasticity in auditory cortex improves neural discrimination of speech sounds.

Authors:  Crystal T Engineer; Jai A Shetake; Navzer D Engineer; Will A Vrana; Jordan T Wolf; Michael P Kilgard
Journal:  Brain Stimul       Date:  2017-01-11       Impact factor: 8.955

Review 3.  Lessons learned from studying syndromic autism spectrum disorders.

Authors:  Yehezkel Sztainberg; Huda Y Zoghbi
Journal:  Nat Neurosci       Date:  2016-10-26       Impact factor: 24.884

4.  Collapse of complexity of brain and body activity due to excessive inhibition and MeCP2 disruption.

Authors:  Jingwen Li; Patrick A Kells; Ayla C Osgood; Shree Hari Gautam; Woodrow L Shew
Journal:  Proc Natl Acad Sci U S A       Date:  2021-10-26       Impact factor: 11.205

5.  Knockdown of Dyslexia-Gene Dcdc2 Interferes with Speech Sound Discrimination in Continuous Streams.

Authors:  Tracy Michelle Centanni; Anne B Booker; Fuyi Chen; Andrew M Sloan; Ryan S Carraway; Robert L Rennaker; Joseph J LoTurco; Michael P Kilgard
Journal:  J Neurosci       Date:  2016-04-27       Impact factor: 6.167

6.  Shank3-deficient rats exhibit degraded cortical responses to sound.

Authors:  Crystal T Engineer; Kimiya C Rahebi; Michael S Borland; Elizabeth P Buell; Kwok W Im; Linda G Wilson; Pryanka Sharma; Sven Vanneste; Hala Harony-Nicolas; Joseph D Buxbaum; Michael P Kilgard
Journal:  Autism Res       Date:  2017-10-20       Impact factor: 5.216

7.  Atypical processing of tones and phonemes in Rett Syndrome as biomarkers of disease progression.

Authors:  Olga V Sysoeva; Sophie Molholm; Aleksandra Djukic; Hans-Peter Frey; John J Foxe
Journal:  Transl Psychiatry       Date:  2020-06-10       Impact factor: 6.222

8.  Impaired Processing in the Primary Auditory Cortex of an Animal Model of Autism.

Authors:  Renata Figueiredo Anomal; Etienne de Villers-Sidani; Juliana Alves Brandão; Rebecca Diniz; Marcos R Costa; Rodrigo N Romcy-Pereira
Journal:  Front Syst Neurosci       Date:  2015-11-16

9.  Characterization of Rett Syndrome-like phenotypes in Mecp2-knockout rats.

Authors:  Yang Wu; Weiwei Zhong; Ningren Cui; Christopher M Johnson; Hao Xing; Shuang Zhang; Chun Jiang
Journal:  J Neurodev Disord       Date:  2016-06-16       Impact factor: 4.025

10.  Loss of MeCP2 in the rat models regression, impaired sociability and transcriptional deficits of Rett syndrome.

Authors:  Surabi Veeraragavan; Ying-Wooi Wan; Daniel R Connolly; Shannon M Hamilton; Christopher S Ward; Sirena Soriano; Meagan R Pitcher; Christopher M McGraw; Sharon G Huang; Jennie R Green; Lisa A Yuva; Agnes J Liang; Jeffrey L Neul; Dag H Yasui; Janine M LaSalle; Zhandong Liu; Richard Paylor; Rodney C Samaco
Journal:  Hum Mol Genet       Date:  2016-06-30       Impact factor: 6.150

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