Acetyl-L-Carnitine Improves Behavior and Dendritic Morphology in a Mouse Model of Rett Syndrome
- Publication Date
- December 05, 2012
- Journal
- PLOS ONE
- Authors
- Laura R. Schaevitz, Raffaella Nicolai, Carla M. Lopez, Stefania D'iddio, et al
- Volume
- 7
- Issue
- 12
- Pages
- e51586
- DOI
- https://dx.plos.org/10.1371/journal.pone.0051586
- Publisher URL
- http://journals.plos.org/plosone/article?id=10.1371%2Fjournal.pone.0051586
- PubMed
- http://www.ncbi.nlm.nih.gov/pubmed/23227269
- PubMed Central
- http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3515484
- Europe PMC
- http://europepmc.org/abstract/MED/23227269
- Web of Science
- 000312588200128
- Scopus
- 84870776384
- Mendeley
- http://www.mendeley.com/research/acetyllcarnitine-improves-behavior-dendritic-morphology-mouse-model-rett-syndrome
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Mendeley | Further Information
{"title"=>"Acetyl-L-Carnitine Improves Behavior and Dendritic Morphology in a Mouse Model of Rett Syndrome", "type"=>"journal", "authors"=>[{"first_name"=>"Laura R.", "last_name"=>"Schaevitz", "scopus_author_id"=>"9435421600"}, {"first_name"=>"Raffaella", "last_name"=>"Nicolai", "scopus_author_id"=>"7003419714"}, {"first_name"=>"Carla M.", "last_name"=>"Lopez", "scopus_author_id"=>"55515623700"}, {"first_name"=>"Stefania", "last_name"=>"D'Iddio", "scopus_author_id"=>"6602930713"}, {"first_name"=>"Emerenziana", "last_name"=>"Iannoni", "scopus_author_id"=>"20734086900"}, {"first_name"=>"Joanne E.", "last_name"=>"Berger-Sweeney", "scopus_author_id"=>"6701330046"}], "year"=>2012, "source"=>"PLoS ONE", "identifiers"=>{"issn"=>"19326203", "scopus"=>"2-s2.0-84870776384", "sgr"=>"84870776384", "pui"=>"366234393", "isbn"=>"1932-6203 (Electronic)\\n1932-6203 (Linking)", "pmid"=>"23227269", "doi"=>"10.1371/journal.pone.0051586"}, "id"=>"bdba739c-7b8b-3b00-a576-7f61936bdcec", "abstract"=>"Rett syndrome (RTT) is a devastating neurodevelopmental disorder affecting 1 in 10,000 girls. Approximately 90% of cases are caused by spontaneous mutations in the X-linked gene encoding methyl-CpG-binding protein 2 (MeCP2). Girls with RTT suffer from severe motor, respiratory, cognitive and social abnormalities attributed to early deficits in synaptic connectivity which manifest in the adult as a myriad of physiological and anatomical abnormalities including, but not limited to, dimished dendritic complexity. Supplementation with acetyl-L-carnitine (ALC), an acetyl group donor, ameliorates motor and cognitive deficits in other disease models through a variety of mechanisms including altering patterns of histone acetylation resulting in changes in gene expression, and stimulating biosynthetic pathways such as acetylcholine. We hypothesized ALC treatment during critical periods in cortical development would promote normal synaptic maturation, and continuing treatment would improve behavioral deficits in the Mecp2(1lox) mouse model of RTT. In this study, wildtype and Mecp2(1lox) mutant mice received daily injections of ALC from birth until death (postnatal day 47). General health, motor, respiratory, and cognitive functions were assessed at several time points during symptom progression. ALC improved weight gain, grip strength, activity levels, prevented metabolic abnormalities and modestly improved cognitive function in Mecp2 null mice early in the course of treatment, but did not significantly improve motor or cognitive functions assessed later in life. ALC treatment from birth was associated with an almost complete rescue of hippocampal dendritic morphology abnormalities with no discernable side effects in the mutant mice. Therefore, ALC appears to be a promising therapeutic approach to treating early RTT symptoms and may be useful in combination with other therapies.", "link"=>"http://www.mendeley.com/research/acetyllcarnitine-improves-behavior-dendritic-morphology-mouse-model-rett-syndrome", "reader_count"=>40, "reader_count_by_academic_status"=>{"Unspecified"=>1, "Professor > Associate Professor"=>2, "Researcher"=>9, "Student > Doctoral Student"=>5, "Student > Ph. D. Student"=>11, "Student > Postgraduate"=>1, "Student > Master"=>8, "Other"=>2, "Professor"=>1}, "reader_count_by_user_role"=>{"Unspecified"=>1, "Professor > Associate Professor"=>2, "Researcher"=>9, "Student > Doctoral Student"=>5, "Student > Ph. D. Student"=>11, "Student > Postgraduate"=>1, "Student > Master"=>8, "Other"=>2, "Professor"=>1}, "reader_count_by_subject_area"=>{"Unspecified"=>3, "Biochemistry, Genetics and Molecular Biology"=>1, "Agricultural and Biological Sciences"=>19, "Medicine and Dentistry"=>11, "Neuroscience"=>1, "Sports and Recreations"=>1, "Pharmacology, Toxicology and Pharmaceutical Science"=>1, "Psychology"=>3}, "reader_count_by_subdiscipline"=>{"Medicine and Dentistry"=>{"Medicine and Dentistry"=>11}, "Neuroscience"=>{"Neuroscience"=>1}, "Sports and Recreations"=>{"Sports and Recreations"=>1}, "Psychology"=>{"Psychology"=>3}, "Agricultural and Biological Sciences"=>{"Agricultural and Biological Sciences"=>19}, "Biochemistry, Genetics and Molecular Biology"=>{"Biochemistry, Genetics and Molecular Biology"=>1}, "Unspecified"=>{"Unspecified"=>3}, "Pharmacology, Toxicology and Pharmaceutical Science"=>{"Pharmacology, Toxicology and Pharmaceutical Science"=>1}}, "reader_count_by_country"=>{"United States"=>1, "China"=>1, "France"=>1}, "group_count"=>0}CrossRef
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Europe PMC Database Citations | Further Information
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Relative Metric
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