Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X Syndrome

Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability due to an expansion in the full mutation range (>200 CGG repeats) of the promoter region of the FMR1 gene leading to gene silencing. Lack of FMRP, a critical protein for dendritic spine formation and maturation...

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Main Authors: Tri Indah Winarni, Andrea Schneider, Mariya Borodyanskara, Randi J. Hagerman
Format: Article
Language:English
Published: Wiley 2012-01-01
Series:Case Reports in Genetics
Online Access:http://dx.doi.org/10.1155/2012/280813
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author Tri Indah Winarni
Andrea Schneider
Mariya Borodyanskara
Randi J. Hagerman
author_facet Tri Indah Winarni
Andrea Schneider
Mariya Borodyanskara
Randi J. Hagerman
author_sort Tri Indah Winarni
collection DOAJ
description Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability due to an expansion in the full mutation range (>200 CGG repeats) of the promoter region of the FMR1 gene leading to gene silencing. Lack of FMRP, a critical protein for dendritic spine formation and maturation, will cause FXS. Early environmental enrichment combined with pharmacological intervention has been proven to rescue dendritic spine abnormalities in the animal model of FXS. Here we report on 2 young children with FXS who were treated early with a combination of targeted treatment and intensive educational interventions leading to improvement in their cognition and behavior and a normal IQ.
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institution Kabale University
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publishDate 2012-01-01
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spelling doaj-art-8b3dad63d119476ea159e9ecb74535f42025-08-20T03:37:34ZengWileyCase Reports in Genetics2090-65442090-65522012-01-01201210.1155/2012/280813280813Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X SyndromeTri Indah Winarni0Andrea Schneider1Mariya Borodyanskara2Randi J. Hagerman3Medical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California at Davis Medical Center, Sacramento, CA 95817, USAMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California at Davis Medical Center, Sacramento, CA 95817, USACollege of Osteopathic Medicine, Touro University California, 1310 Johnson Lane, Mare Island, Vallejo, CA 94592, USAMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California at Davis Medical Center, Sacramento, CA 95817, USAFragile X syndrome (FXS) is the most common inherited cause of intellectual disability due to an expansion in the full mutation range (>200 CGG repeats) of the promoter region of the FMR1 gene leading to gene silencing. Lack of FMRP, a critical protein for dendritic spine formation and maturation, will cause FXS. Early environmental enrichment combined with pharmacological intervention has been proven to rescue dendritic spine abnormalities in the animal model of FXS. Here we report on 2 young children with FXS who were treated early with a combination of targeted treatment and intensive educational interventions leading to improvement in their cognition and behavior and a normal IQ.http://dx.doi.org/10.1155/2012/280813
spellingShingle Tri Indah Winarni
Andrea Schneider
Mariya Borodyanskara
Randi J. Hagerman
Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X Syndrome
Case Reports in Genetics
title Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X Syndrome
title_full Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X Syndrome
title_fullStr Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X Syndrome
title_full_unstemmed Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X Syndrome
title_short Early Intervention Combined with Targeted Treatment Promotes Cognitive and Behavioral Improvements in Young Children with Fragile X Syndrome
title_sort early intervention combined with targeted treatment promotes cognitive and behavioral improvements in young children with fragile x syndrome
url http://dx.doi.org/10.1155/2012/280813
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