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Research
Katherine S. Adcock
University of Texas at Dallas
Corresponding Author
ORCiD: https://orcid.org/0000-0002-8527-7396
License:
This work is licensed under a CC BY 4.0 License. Read Full License
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.
Keywords
Rett syndrome, MeCP2, motor, auditory, learning
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Peer Review Timeline
CURRENT STATUS: Published
View the published article at Journal of Neurodevelopmental Disorders.
No community comments so far
Editorial decision: Accept
On 17 Sep, 2020
Review #1 received
Received 15 Sep, 2020
Editor assigned
On 01 Sep, 2020
Reviewers invited
Invitations sent on 01 Sep, 2020
Reviewer #1 agreed
On 01 Sep, 2020
Submission checks complete
On 31 Aug, 2020
Editor invited
On 31 Aug, 2020
No comments provided
Editorial decision: Major revision
On 12 Aug, 2020
Review #2 received
Received 07 Aug, 2020
Review #1 received
Received 04 Aug, 2020
Reviewer #2 agreed
On 24 Jul, 2020
Editor assigned
On 23 Jul, 2020
Reviewers invited
Invitations sent on 23 Jul, 2020
Reviewer #1 agreed
On 23 Jul, 2020
Submission checks complete
On 22 Jul, 2020
Editor invited
On 22 Jul, 2020
Version 1
Posted 02 Jun, 2020
No comments provided
Editorial decision: Major revision
On 23 Jun, 2020
Review #2 received
Received 22 Jun, 2020
Review #1 received
Received 10 Jun, 2020
Reviewer #2 agreed
On 06 Jun, 2020
Reviewer #3 agreed
On 06 Jun, 2020
Reviewer #1 agreed
On 06 Jun, 2020
Reviewers invited
Invitations sent on 03 Jun, 2020
First submitted
On 26 May, 2020
Editor assigned
On 26 May, 2020
Submission checks complete
On 25 May, 2020
Editor invited
On 25 May, 2020
Metrics
Comments: 0
PDF Downloads: ...
HTML Views: ...
Subject Areas
Developmental Neuroscience
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A new preprint design is coming!
We have improved readability, clarity, accessibility, and opportunities for engagement.
See the published version of this preprint at Journal of Neurodevelopmental Disorders.
This is a preprint, a preliminary version of a manuscript that has not completed peer review at a journal. Research Square does not conduct peer review prior to posting preprints. The posting of a preprint on this server should not be interpreted as an endorsement of its validity or suitability for dissemination as established information or for guiding clinical practice.
Learn more about In Review
Research
Katherine S. Adcock
University of Texas at Dallas
Corresponding Author
ORCiD: https://orcid.org/0000-0002-8527-7396
Badges
Prescreen
This manuscript passed our Prescreen assessment
Complete author information
Appropriate declaration statements
Potential risks to human health
Prescreen
Peer Review Timeline
CURRENT STATUS: Published
View the published article at Journal of Neurodevelopmental Disorders.
No community comments so far
Editorial decision: Accept
On 17 Sep, 2020
Review #1 received
Received 15 Sep, 2020
Editor assigned
On 01 Sep, 2020
Reviewers invited
Invitations sent on 01 Sep, 2020
Reviewer #1 agreed
On 01 Sep, 2020
Submission checks complete
On 31 Aug, 2020
Editor invited
On 31 Aug, 2020
No comments provided
Editorial decision: Major revision
On 12 Aug, 2020
Review #2 received
Received 07 Aug, 2020
Review #1 received
Received 04 Aug, 2020
Reviewer #2 agreed
On 24 Jul, 2020
Editor assigned
On 23 Jul, 2020
Reviewers invited
Invitations sent on 23 Jul, 2020
Reviewer #1 agreed
On 23 Jul, 2020
Submission checks complete
On 22 Jul, 2020
Editor invited
On 22 Jul, 2020
Version 1
Posted 02 Jun, 2020
No comments provided
Editorial decision: Major revision
On 23 Jun, 2020
Review #2 received
Received 22 Jun, 2020
Review #1 received
Received 10 Jun, 2020
Reviewer #2 agreed
On 06 Jun, 2020
Reviewer #3 agreed
On 06 Jun, 2020
Reviewer #1 agreed
On 06 Jun, 2020
Reviewers invited
Invitations sent on 03 Jun, 2020
First submitted
On 26 May, 2020
Editor assigned
On 26 May, 2020
Submission checks complete
On 25 May, 2020
Editor invited
On 25 May, 2020
Metrics
Comments: 0
PDF Downloads: ...
HTML Views: ...
Subject Areas
Developmental Neuroscience
License:
This work is licensed under a CC BY 4.0 License. Read Full License
View the published article at Journal of Neurodevelopmental Disorders.
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.
Figure 1
Figure 2
Figure 3
Figure 4
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