Literature DB >> 18321865

Defective body-weight regulation, motor control and abnormal social interactions in Mecp2 hypomorphic mice.

Bredford Kerr1, Matías Alvarez-Saavedra, Mauricio A Sáez, Alexandra Saona, Juan I Young.   

Abstract

MeCP2 is an abundant protein that binds to methylated cytosine residues in DNA and regulates transcription. Mutations in MECP2 cause Rett syndrome, a severe neurological disorder that affects approximately 1:10 000 females. Mice lacking MeCP2 have been generated and constitute important models of Rett syndrome. However, it is yet unclear whether certain physiological events are sensitive to a decrease, rather than a complete lack of MeCP2. Here we report that a Mecp2 floxed allele (Mecp2(lox)) that was generated to allow conditional mutagenesis behaves as a hypomorph and the corresponding mutant mice exhibit phenotypical alterations including body weight gain, motor abnormalities and altered social behavior. Our data reinforce the view that the central nervous system is extremely sensitive to MeCP2 expression levels and suggest that the 3'-UTR of Mecp2 might contain important elements that contribute to the regulation of its stability or processing.

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Year:  2008        PMID: 18321865     DOI: 10.1093/hmg/ddn061

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  44 in total

Review 1.  Altered trajectories of neurodevelopment and behavior in mouse models of Rett syndrome.

Authors:  Elizabeth S Smith; Dani R Smith; Charlotte Eyring; Maria Braileanu; Karen S Smith-Connor; Yew Ei Tan; Amanda Y Fowler; Gloria E Hoffman; Michael V Johnston; Sujatha Kannan; Mary E Blue
Journal:  Neurobiol Learn Mem       Date:  2018-11-29       Impact factor: 2.877

2.  Loss of MeCP2 in cholinergic neurons causes part of RTT-like phenotypes via α7 receptor in hippocampus.

Authors:  Ying Zhang; Shu-Xia Cao; Peng Sun; Hai-Yang He; Ci-Hang Yang; Xiao-Juan Chen; Chen-Jie Shen; Xiao-Dong Wang; Zhong Chen; Darwin K Berg; Shumin Duan; Xiao-Ming Li
Journal:  Cell Res       Date:  2016-04-22       Impact factor: 25.617

3.  MeCP2 isoform e1 mutant mice recapitulate motor and metabolic phenotypes of Rett syndrome.

Authors:  Annie Vogel Ciernia; Dag H Yasui; Michael C Pride; Blythe Durbin-Johnson; Adriana B Noronha; Alene Chang; Trina A Knotts; Jennifer R Rutkowsky; Jon J Ramsey; Jacqueline N Crawley; Janine M LaSalle
Journal:  Hum Mol Genet       Date:  2018-12-01       Impact factor: 6.150

4.  Mice with an isoform-ablating Mecp2 exon 1 mutation recapitulate the neurologic deficits of Rett syndrome.

Authors:  Dag H Yasui; Michael L Gonzales; Justin O Aflatooni; Florence K Crary; Daniel J Hu; Bryant J Gavino; Mari S Golub; John B Vincent; N Carolyn Schanen; Carl O Olson; Mojgan Rastegar; Janine M Lasalle
Journal:  Hum Mol Genet       Date:  2013-12-18       Impact factor: 6.150

5.  MeCP2 reinforces STAT3 signaling and the generation of effector CD4+ T cells by promoting miR-124-mediated suppression of SOCS5.

Authors:  Shan Jiang; Chaoran Li; Gabrielle McRae; Erik Lykken; Jose Sevilla; Si-Qi Liu; Ying Wan; Qi-Jing Li
Journal:  Sci Signal       Date:  2014-03-11       Impact factor: 8.192

Review 6.  Leveraging the genetic basis of Rett syndrome to ascertain pathophysiology.

Authors:  Hua Yang; Kequan Li; Song Han; Ailing Zhou; Zhaolan Joe Zhou
Journal:  Neurobiol Learn Mem       Date:  2018-11-14       Impact factor: 2.877

7.  Transgenic complementation of MeCP2 deficiency: phenotypic rescue of Mecp2-null mice by isoform-specific transgenes.

Authors:  Bredford Kerr; Jessica Soto C; Mauricio Saez; Alexander Abrams; Katherina Walz; Juan I Young
Journal:  Eur J Hum Genet       Date:  2011-08-10       Impact factor: 4.246

Review 8.  Rett syndrome: a complex disorder with simple roots.

Authors:  Matthew J Lyst; Adrian Bird
Journal:  Nat Rev Genet       Date:  2015-03-03       Impact factor: 53.242

9.  A Role for Diminished GABA Transporter Activity in the Cortical Discharge Phenotype of MeCP2-Deficient Mice.

Authors:  Liang Zhang; Robert G Wither; Min Lang; Chiping Wu; Elena Sidorova-Darmos; Hristo Netchev; Catherine B Matolcsy; Orlando Carter Snead; James H Eubanks
Journal:  Neuropsychopharmacology       Date:  2015-10-26       Impact factor: 7.853

10.  A novel hypomorphic MECP2 point mutation is associated with a neuropsychiatric phenotype.

Authors:  Abidemi A Adegbola; Michael L Gonzales; Andrew Chess; Janine M LaSalle; Gerald F Cox
Journal:  Hum Genet       Date:  2008-11-07       Impact factor: 4.132

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