Literature DB >> 10208160

Electrodermal responses to sensory stimuli in individuals with fragile X syndrome: a preliminary report.

L J Miller1, D N McIntosh, J McGrath, V Shyu, M Lampe, A K Taylor, F Tassone, K Neitzel, T Stackhouse, R J Hagerman.   

Abstract

The fragile X mutation and fragile X syndrome are associated with hyperarousal, hyperactivity, aggression, and anxiety. These may be related to strong reactions to auditory, tactile, visual, and olfactory stimuli [Hagerman, 1996b; Hagerman and Cronister, 1996]. However, almost no data exist describing hyperarousal and sensory sensitivity in individuals with the fragile X mutation. This study establishes a reliable laboratory paradigm for examining reactions to sensory stimuli. We found the pattern of electrodermal responses (EDRs) to stimulation in one sensory modality predicted the pattern of EDRs in four other sensory systems. In addition, the EDR pattern of individuals with the fragile X mutation was related to their FMR-protein expression. Finally, EDRs in individuals with fragile X syndrome were significantly different from those of normal controls, demonstrating greater magnitude, more responses per stimulation, responses on a greater proportion of trials, and lower rates of habituation. The findings support the theory that individuals with fragile X syndrome have a physiologically based enhancement of reactions to sensations. Because electrodermal activity indexes sympathetic nervous system activity, the data suggest that the over-arousal to sensation may involve the sympathetic system.

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Year:  1999        PMID: 10208160

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  140 in total

Review 1.  Fragile X syndrome: the GABAergic system and circuit dysfunction.

Authors:  Scott M Paluszkiewicz; Brandon S Martin; Molly M Huntsman
Journal:  Dev Neurosci       Date:  2011-09-21       Impact factor: 2.984

Review 2.  Fragile X: leading the way for targeted treatments in autism.

Authors:  Lulu W Wang; Elizabeth Berry-Kravis; Randi J Hagerman
Journal:  Neurotherapeutics       Date:  2010-07       Impact factor: 7.620

Review 3.  Fragile X syndrome and targeted treatment trials.

Authors:  Randi Hagerman; Julie Lauterborn; Jacky Au; Elizabeth Berry-Kravis
Journal:  Results Probl Cell Differ       Date:  2012

4.  The GABA(A) receptor agonist THIP ameliorates specific behavioral deficits in the mouse model of fragile X syndrome.

Authors:  Jose Luis Olmos-Serrano; Joshua G Corbin; Mark P Burns
Journal:  Dev Neurosci       Date:  2011-11-08       Impact factor: 2.984

5.  Potential Involvement of Impaired BKCa Channel Function in Sensory Defensiveness and Some Behavioral Disturbances Induced by Unfamiliar Environment in a Mouse Model of Fragile X Syndrome.

Authors:  Maria Isabel Carreno-Munoz; Fabienne Martins; Maria Carmen Medrano; Elisabetta Aloisi; Susanna Pietropaolo; Corentin Dechaud; Enejda Subashi; Guillaume Bony; Melanie Ginger; Abdelmalik Moujahid; Andreas Frick; Xavier Leinekugel
Journal:  Neuropsychopharmacology       Date:  2017-07-19       Impact factor: 7.853

6.  Physiological arousal in autism and fragile X syndrome: group comparisons and links with pragmatic language.

Authors:  Jessica Klusek; Gary E Martin; Molly Losh
Journal:  Am J Intellect Dev Disabil       Date:  2013-11

7.  Fragile X mental retardation protein regulates olfactory sensitivity but not odorant discrimination.

Authors:  Arielle Schilit Nitenson; Emily E Stackpole; Torrey L S Truszkowski; Maellie Midroit; Justin R Fallon; Kevin G Bath
Journal:  Chem Senses       Date:  2015-04-27       Impact factor: 3.160

8.  National survey of sensory features in children with ASD: factor structure of the sensory experience questionnaire (3.0).

Authors:  Karla Ausderau; John Sideris; Melissa Furlong; Lauren M Little; John Bulluck; Grace T Baranek
Journal:  J Autism Dev Disord       Date:  2014-04

Review 9.  Neuropsychiatric symptoms of fragile X syndrome: pathophysiology and pharmacotherapy.

Authors:  John A Tsiouris; W Ted Brown
Journal:  CNS Drugs       Date:  2004       Impact factor: 5.749

Review 10.  Fragile X: a family of disorders.

Authors:  Weerasak Chonchaiya; Andrea Schneider; Randi J Hagerman
Journal:  Adv Pediatr       Date:  2009
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