Literature DB >> 28544139

RettBASE: Rett syndrome database update.

Rahul Krishnaraj1, Gladys Ho1,2, John Christodoulou1,2,3,4.   

Abstract

Rett syndrome (RTT) is an X-linked progressive neurodevelopmental disorder that primarily affects females. Mutations in the MECP2 gene have been attributed as the major genetic cause of RTT. Recently, mutations in CDKL5 and FOXG1 genes have also been suggested to give rise to RTT, although subsequent more extensive studies suggest that diseases resulting from mutations in these two genes should be considered as distinct clinical entities. While the genetic basis for the RTT has been recognized, so far there is no effective cure for the disease and the treatments available are mainly aimed at ameliorating clinical problems associated with the disorder. The swift identification of the mutations in children is crucial for pursuing the best therapeutic care. RettBASE was created in 2002 as a MECP2 variant database and has grown to become a comprehensive variant database for RTT and related clinical phenotypes, containing a curated collection of variants for MECP2, CDKL5, and FOXG1 genes. Here, we describe the development and growth of RettBASE after its inception in 2001. Currently, RettBASE holds a total of 4,668 variants in MECP2, 498 variants in CDKL5, and 64 variants in FOXG1.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  CDKL5; FOXG1; MECP2; Rett syndrome; database; locus-specific database; mutation; phenotype-genotype correlations

Mesh:

Substances:

Year:  2017        PMID: 28544139     DOI: 10.1002/humu.23263

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  38 in total

1.  Dysregulation of BRD4 Function Underlies the Functional Abnormalities of MeCP2 Mutant Neurons.

Authors:  Yangfei Xiang; Yoshiaki Tanaka; Benjamin Patterson; Sung-Min Hwang; Eriona Hysolli; Bilal Cakir; Kun-Yong Kim; Wanshan Wang; Young-Jin Kang; Ethan M Clement; Mei Zhong; Sang-Hun Lee; Yee Sook Cho; Prabir Patra; Gareth J Sullivan; Sherman M Weissman; In-Hyun Park
Journal:  Mol Cell       Date:  2020-06-10       Impact factor: 17.970

2.  Neurobiology: Domains to the rescue for Rett syndrome.

Authors:  Anne E West
Journal:  Nature       Date:  2017-10-11       Impact factor: 49.962

3.  Surface- and voxel-based brain morphologic study in Rett and Rett-like syndrome with MECP2 mutation.

Authors:  Tadashi Shiohama; Jacob Levman; Emi Takahashi
Journal:  Int J Dev Neurosci       Date:  2019-01-25       Impact factor: 2.457

4.  Loss of O-GlcNAcylation on MeCP2 at Threonine 203 Leads to Neurodevelopmental Disorders.

Authors:  Juanxian Cheng; Zhe Zhao; Liping Chen; Ying Li; Ruijing Du; Yan Wu; Qian Zhu; Ming Fan; Xiaotao Duan; Haitao Wu
Journal:  Neurosci Bull       Date:  2021-11-12       Impact factor: 5.203

5.  Adenine Base Editing In Vivo with a Single Adeno-Associated Virus Vector.

Authors:  Han Zhang; Nathan Bamidele; Pengpeng Liu; Ogooluwa Ojelabi; Xin D Gao; Tomás Rodriguez; Haoyang Cheng; Karen Kelly; Jonathan K Watts; Jun Xie; Guangping Gao; Scot A Wolfe; Wen Xue; Erik J Sontheimer
Journal:  GEN Biotechnol       Date:  2022-06-14

6.  Combined in Silico Prediction Methods, Molecular Dynamic Simulation, and Molecular Docking of FOXG1 Missense Mutations: Effect on FoxG1 Structure and Its Interactions with DNA and Bmi-1 Protein.

Authors:  Marwa Kharrat; Chahnez Charfi Triki; Olfa Alila-Fersi; Olfa Jallouli; Bassem Khemakham; Salma Mallouli; Marwa Maalej; Marwa Ammar; Fakher Frikha; Fatma Kamoun; Faiza Fakhfakh
Journal:  J Mol Neurosci       Date:  2022-06-02       Impact factor: 2.866

7.  Recommendations by the ClinGen Rett/Angelman-like expert panel for gene-specific variant interpretation methods.

Authors:  Dianalee McKnight; Lora Bean; Izabela Karbassi; Katelynn Beattie; Thierry Bienvenu; Hope Bonin; Ping Fang; John Chrisodoulou; Michael Friez; Maria Helgeson; Rahul Krishnaraj; Linyan Meng; Lindsey Mighion; Jeffrey Neul; Alan Percy; Simon Ramsden; Huda Zoghbi; Soma Das
Journal:  Hum Mutat       Date:  2021-12-02       Impact factor: 4.700

8.  Expression pattern of cdkl5 during zebrafish early development: implications for use as model for atypical Rett syndrome.

Authors:  Marta Vitorino; Nídia Cunha; Natércia Conceição; M Leonor Cancela
Journal:  Mol Biol Rep       Date:  2018-05-11       Impact factor: 2.316

9.  Rett syndrome-causing mutations compromise MeCP2-mediated liquid-liquid phase separation of chromatin.

Authors:  Liang Wang; Mingli Hu; Mei-Qing Zuo; Jicheng Zhao; Di Wu; Li Huang; Yongxin Wen; Yunfan Li; Ping Chen; Xinhua Bao; Meng-Qiu Dong; Guohong Li; Pilong Li
Journal:  Cell Res       Date:  2020-02-28       Impact factor: 25.617

Review 10.  The distinct methylation landscape of maturing neurons and its role in Rett syndrome pathogenesis.

Authors:  Laura A Lavery; Huda Y Zoghbi
Journal:  Curr Opin Neurobiol       Date:  2019-09-19       Impact factor: 6.627

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