نتایج جستجو برای: fxs

تعداد نتایج: 581  

2013
Travis L Schmit James A Dowell Margaret E Maes Michael Wilhelm

Fragile X syndrome (FXS) is caused by the loss of functional fragile X mental retardation protein (FMRP). Loss of FMRP results in an elevated basal protein expression profile of FMRP targeted mRNAs, a loss of local metabotropic glutamate receptor (mGluR)-regulated protein synthesis, exaggerated long-term depression and corresponding learning and behavioral deficits. Evidence shows that blocking...

Journal: :Mental retardation and developmental disabilities research reviews 2004
David Hessl Susan M Rivera Allan L Reiss

Fragile X syndrome (FXS), caused by a single gene mutation on the X chromosome, offers a unique opportunity for investigation of gene-brain-behavior relationships. Recent advances in molecular genetics, human brain imaging, and behavioral studies have started to unravel the complex pathways leading to the cognitive, psychiatric, and physical features that are unique to this syndrome. In this ar...

2017
Verónica Martínez-Cerdeño Mirna Lechpammer Stephen Noctor Jeanelle Ariza Paul Hagerman Randi Hagerman

This is a report of FMR1 premutation with Prader-Willi phenotype (PWP) and FXTAS. Although the PWP is common in fragile X syndrome (FXS), it has never been described in someone with the premutation. The patient presented intranuclear inclusions, severe obesity, hyperphagia, and ADHD symptoms, typical of the PWP in FXS. In addition, the autopsy revealed multiple architectural cortical abnormalit...

2017
Chariyawan Charalsawadi Juthamas Wirojanan Somchit Jaruratanasirikul Nichara Ruangdaraganon Alan Geater Pornprot Limprasert

Background. Clinical characteristics of fragile X syndrome (FXS) have been well documented in Caucasians, whereas in Asians they have rarely been described. Those that have been conducted used small cohorts that utilized DNA for diagnosis and larger cohorts that utilized cytogenetics for diagnosis. This study is to describe clinical characteristics of FXS in a large cohort of Thai patients diag...

2016
Jantine A. C. Broek Zhanmin Lin H. Martijn de Gruiter Heleen van ‘t Spijker Elize D. Haasdijk David Cox Sureyya Ozcan Gert W. A. van Cappellen Adriaan B. Houtsmuller Rob Willemsen Chris I. de Zeeuw Sabine Bahn

BACKGROUND Fragile X syndrome (FXS) is a single-gene disorder that is the most common heritable cause of intellectual disability and the most frequent monogenic cause of autism spectrum disorders (ASD). FXS is caused by an expansion of trinucleotide repeats in the promoter region of the fragile X mental retardation gene (Fmr1). This leads to a lack of fragile X mental retardation protein (FMRP)...

Journal: :American journal of medical genetics. Part A 2008
M Giulia Torrioli Silvia Vernacotola Laura Peruzzi Elisabetta Tabolacci Montserrat Mila Roberto Militerni Sebastiano Musumeci Feliciano J Ramos Marìa Frontera Giovanni Sorge Elisabetta Marzullo Giusi Romeo Louis Vallee Edvige Veneselli Elena Cocchi Eleonora Garbarino Umberto Moscato Pietro Chiurazzi Stefania D'Iddio Menotti Calvani Giovanni Neri

Attention deficit hyperactivity disorder (ADHD) is a frequent behavioral problem in young boys with fragile X syndrome (FXS), and its treatment is critical for improving social ability. The short-term efficacy of stimulant medications like methylphenidate (MPH) is well established in children with ADHD. FXS boys treated with MPH have improved attention span and socialization skills; however the...

Journal: :American journal of medical genetics. Part C, Seminars in medical genetics 2015
Lisa Cochran Joanna Moss Lisa Nelson Chris Oliver

Little is known about the way in which the characteristics of autism spectrum disorder (ASD) develop and manifest across the age span in individuals with genetic syndromes. In this study we present findings from a two and a half year follow-up of the characteristics associated with ASD in three syndromes: Cornelia de Lange (CdLS), Fragile X (FXS), and Cri du Chat (CdCS). Parents and carers of 2...

Journal: :Human brain mapping 2014
David Romano Monica Nicolau Eve-Marie Quintin Paul K Mazaika Amy A Lightbody Heather Cody Hazlett Joseph Piven Gunnar Carlsson Allan L Reiss

Fragile X syndrome (FXS), due to mutations of the FMR1 gene, is the most common known inherited cause of developmental disability as well as the most common single-gene risk factor for autism. Our goal was to examine variation in brain structure in FXS with topological data analysis (TDA), and to assess how such variation is associated with measures of IQ and autism-related behaviors. To this e...

2014
Dalyir Pretto Carolyn M. Yrigollen Hiu-Tung Tang John Williamson Glenda Espinal Chris K. Iwahashi Blythe Durbin-Johnson Randi J. Hagerman Paul J. Hagerman Flora Tassone

Expansions of more than 200 CGG repeats (full mutation) in the FMR1 gene give rise to fragile X syndrome (FXS) through a process that generally involves hypermethylation of the FMR1 promoter region and gene silencing, resulting in absence of expression of the encoded protein, FMRP. However, mosaicism with alleles differing in size and extent of methylation often exist within or between tissues ...

2017
Shimriet Zeidler Helen de Boer Renate K. Hukema Rob Willemsen

Fragile X syndrome (FXS) is the most common monogenetic cause of intellectual disability and autism. The disorder is characterized by altered synaptic plasticity in the brain. Synaptic plasticity is tightly regulated by a complex balance of different synaptic pathways. In FXS, various synaptic pathways are disrupted, including the excitatory metabotropic glutamate receptor 5 (mGluR5) and the in...

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