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      Repetitive and self-injurious behaviors: associations with caudate volume in autism and fragile X syndrome

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          Abstract

          Background

          Following from previous work suggesting that neurobehavioral features distinguish fragile X and idiopathic variants of autism, we investigated the relationships between four forms of repetitive behavior (stereotypy, self-injury, compulsivity, ritual behavior) and caudate nuclei volume in two groups: boys with fragile X syndrome, a subset of whom met criteria for autism, and a comparison group of boys with idiopathic autism.

          Methods

          Bilateral caudate nuclei volumes were measured in boys aged 3 to 6 years with fragile X syndrome ( n = 41), the subset of boys with fragile X syndrome and autism ( n = 16), and boys with idiopathic autism ( n = 30). Repetitive behaviors were measured using the Repetitive Behavior Scales-Revised.

          Results

          For boys with idiopathic autism, left caudate volume was modestly associated with self-injury, while both compulsive and ritual behaviors showed significant positive correlations with bilateral caudate nuclei volumes, replicating previous results. For boys with fragile X syndrome, there was no such association between caudate volume and compulsive behaviors. However, we did identify significant positive correlations between self-injury total scores and number of self-injury topographies with bilateral caudate nuclei volumes.

          Conclusions

          These findings suggest a specific role for the caudate nucleus in the early pathogenesis of self-injurious behavior associated with both idiopathic autism and fragile X syndrome. Results further indicate that the caudate may be differentially associated with compulsive behavior, highlighting the utility of isolating discrete brain-behavior associations within and between subtypes of autism spectrum disorder.

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          Most cited references39

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          Synaptic density in human frontal cortex - developmental changes and effects of aging.

          Density of synaptic profiles in layer 3 of middle frontal gyrus was quantitated in 21 normal human brains ranging from newborn to age 90 years. Synaptic profiles could be reliably demonstrated by the phosphotungstic acid method (Bloom and Aghajanian) in tissue fixed up to 36 h postmortem. Synaptic density was constant throughout adult life (ages 16--72 years) with a mean of 11.05 X 10(8) synapses/cu.mm +/- 0.41 S.E.M. There was a slight decline in synaptic density in brains of the aged (ages 74--90 years) with a mean of 9.56 X 10(8) synapses/cu.mm +/- 0.28 S.E.M. in 4 samples (P less than 0.05). Synaptic density in neonatal brains was already high--in the range seen in adults. However, synaptic morphology differed; immature profiles had an irregular presynaptic dense band instead of the separate presynaptic projections seen in mature synapses. Synaptic density increased during infancy, reaching a maximum at age 1--2 years which was about 50% above the adult mean. The decline in synaptic density observed between ages 2--16 years was accompanied by a slight decrease in neuronal density. Human cerebral cortex is one of a number of neuronal systems in which loss of neurons and synapses appears to occur as a late developmental event.
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            Varieties of repetitive behavior in autism: comparisons to mental retardation.

            Systematic study of abnormal repetitive behaviors in autism has been lacking despite the diagnostic significance of such behavior. The occurrence of specific topographies of repetitive behaviors as well as their severity was assessed in individuals with mental retardation with and without autism. The occurrence of each behavior category, except dyskinesias, was higher in the autism group and autistic subjects exhibited a significantly greater number of topographies of stereotypy and compulsions. Both groups had significant patterns of repetitive behavior co-occurrence. Autistic subjects had significantly greater severity ratings for compulsions, stereotypy, and self-injury. Repetitive behavior severity also predicted severity of autism. Although abnormal repetition is not specific to autism, an elevated pattern of occurrence and severity appears to characterize the disorder.
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              Chronic pharmacological mGlu5 inhibition corrects fragile X in adult mice.

              Fragile X syndrome (FXS) is the most common form of inherited intellectual disability. Previous studies have implicated mGlu5 in the pathogenesis of the disease, but a crucial unanswered question is whether pharmacological mGlu5 inhibition is able to reverse an already established FXS phenotype in mammals. Here we have used the novel, potent, and selective mGlu5 inhibitor CTEP to address this issue in the Fmr1 knockout mouse. Acute CTEP treatment corrects elevated hippocampal long-term depression, protein synthesis, and audiogenic seizures. Chronic treatment that inhibits mGlu5 within a receptor occupancy range of 81% ± 4% rescues cognitive deficits, auditory hypersensitivity, aberrant dendritic spine density, overactive ERK and mTOR signaling, and partially corrects macroorchidism. This study shows that a comprehensive phenotype correction in FXS is possible with pharmacological intervention starting in young adulthood, after development of the phenotype. It is of great interest how these findings may translate into ongoing clinical research testing mGlu5 inhibitors in FXS patients. Copyright © 2012 Elsevier Inc. All rights reserved.
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                Author and article information

                Journal
                J Neurodev Disord
                J Neurodev Disord
                Journal of Neurodevelopmental Disorders
                BioMed Central
                1866-1947
                1866-1955
                2013
                2 May 2013
                : 5
                : 1
                : 12
                Affiliations
                [1 ]Carolina Institute for Developmental Disabilities, University of North Carolina at Chapel Hill School of Medicine, CB# 3367, Chapel Hill, NC, 27599, USA
                [2 ]Department of Psychiatry, University of North Carolina at Chapel Hill School of Medicine, CB# 7160, Chapel Hill, NC, 27599, USA
                [3 ]Center for Interdisciplinary Brain Science Research, Stanford University, 401 Quarry Rd., MC 5795, Stanford, CA, 94305, USA
                [4 ]Department of Psychiatry and Behavioral Sciences, Stanford University, 401 Quarry Rd., MC 5717, Stanford, CA, 94305, USA
                Article
                1866-1955-5-12
                10.1186/1866-1955-5-12
                3651404
                23639144
                304d01f0-f912-4285-b7e1-9f64b7701762
                Copyright ©2013 Wolff et al.; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 24 January 2013
                : 17 April 2013
                Categories
                Research

                Neurosciences
                autism,caudate,fragile x syndrome,repetitive behavior,self-injurious behavior
                Neurosciences
                autism, caudate, fragile x syndrome, repetitive behavior, self-injurious behavior

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