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Unilateral Rhythmic Hand Tapping in Rett Syndrome: Is This Stereotypy?
Rett syndrome is a severe neurodevelopmental disorder, with hand stereotypies as a hallmark of the disease. Epilepsy is a frequent comorbidity and is accompanied by centrotemporal spikes on electroencephalogram, but stereotypic movements should not have epileptiform correlates. During routine video-...
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Published in: | Journal of child neurology 2013-10, Vol.28 (10), p.1210-1214 |
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description | Rett syndrome is a severe neurodevelopmental disorder, with hand stereotypies as a hallmark of the disease. Epilepsy is a frequent comorbidity and is accompanied by centrotemporal spikes on electroencephalogram, but stereotypic movements should not have epileptiform correlates. During routine video-electroencephalographic investigation in 5 Rett syndrome patients, we identified a peculiar type of unilateral, highly rhythmic hand tapping accompanied by contralateral synchronous centrotemporal spikes on electroencephalography. This phenomenon is not consistent with either reflex seizures or hand stereotypies and does not respond to antiepileptic drugs. The electroencephalographic activity probably represents evoked potentials, either somatosensory or motor, whereas the rhythmic activity raises the possibility of a subcortical pacemaker for a stereotypy variant. The phenomenon could be caused by abnormal circuitry among the hyperexcitable somatosensory cortex, motor cortex, and subcortical areas in Rett syndrome. Clinicians should be aware of the nonepileptic nature of this motor behavior and should not attempt to treat it. |
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Epilepsy is a frequent comorbidity and is accompanied by centrotemporal spikes on electroencephalogram, but stereotypic movements should not have epileptiform correlates. During routine video-electroencephalographic investigation in 5 Rett syndrome patients, we identified a peculiar type of unilateral, highly rhythmic hand tapping accompanied by contralateral synchronous centrotemporal spikes on electroencephalography. This phenomenon is not consistent with either reflex seizures or hand stereotypies and does not respond to antiepileptic drugs. The electroencephalographic activity probably represents evoked potentials, either somatosensory or motor, whereas the rhythmic activity raises the possibility of a subcortical pacemaker for a stereotypy variant. The phenomenon could be caused by abnormal circuitry among the hyperexcitable somatosensory cortex, motor cortex, and subcortical areas in Rett syndrome. 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Clinicians should be aware of the nonepileptic nature of this motor behavior and should not attempt to treat it.</description><subject>Cerebral Cortex - physiopathology</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Electroencephalography</subject><subject>Evoked Potentials - physiology</subject><subject>Female</subject><subject>Hand</subject><subject>Humans</subject><subject>Periodicity</subject><subject>Rett Syndrome - physiopathology</subject><subject>Stereotyped Behavior - physiology</subject><issn>0883-0738</issn><issn>1708-8283</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2013</creationdate><recordtype>article</recordtype><recordid>eNqNkM9Lw0AQhRdRbK3ePUmOXqKzO9vs5ihFrVAQansOm82kTckvd5ND_3tTWj0Igqc5vO89mI-xWw4PnCv1CFojKNRcyKlEFGdszBXoUAuN52x8iMNDPmJX3u8AQE9juGQjIWIuUcVjxtd1UZqOnCmD5XbfbavCBnNTZ8HKtG1Rb4KiDpbUdcHHvs5cU9E1u8hN6enmdCds_fK8ms3Dxfvr2-xpEVoU2IUmgjRF4JFUGHFJRHqKGaGOI02UkraWW6GssMhzwSGzWW5BaqlTQGEIJ-z-uNu65rMn3yVV4S2Vpamp6X0yPBALHkdK_gdFoQSXekDhiFrXeO8oT1pXVMbtEw7JwWny2-lQuTut92lF2U_hW-IAhEfAmw0lu6Z39SDm78EvNPx8HQ</recordid><startdate>201310</startdate><enddate>201310</enddate><creator>Nissenkorn, A.</creator><creator>Ben-Zeev, B.</creator><general>SAGE Publications</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>7TK</scope></search><sort><creationdate>201310</creationdate><title>Unilateral Rhythmic Hand Tapping in Rett Syndrome</title><author>Nissenkorn, A. ; Ben-Zeev, B.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c323t-a60bb3016473614eee853de38968eebe8cc1c27c2c31f210dcdfc04848b032ae3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2013</creationdate><topic>Cerebral Cortex - physiopathology</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Electroencephalography</topic><topic>Evoked Potentials - physiology</topic><topic>Female</topic><topic>Hand</topic><topic>Humans</topic><topic>Periodicity</topic><topic>Rett Syndrome - physiopathology</topic><topic>Stereotyped Behavior - physiology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Nissenkorn, A.</creatorcontrib><creatorcontrib>Ben-Zeev, B.</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>Neurosciences Abstracts</collection><jtitle>Journal of child neurology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Nissenkorn, A.</au><au>Ben-Zeev, B.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Unilateral Rhythmic Hand Tapping in Rett Syndrome: Is This Stereotypy?</atitle><jtitle>Journal of child neurology</jtitle><addtitle>J Child Neurol</addtitle><date>2013-10</date><risdate>2013</risdate><volume>28</volume><issue>10</issue><spage>1210</spage><epage>1214</epage><pages>1210-1214</pages><issn>0883-0738</issn><eissn>1708-8283</eissn><notes>ObjectType-Article-1</notes><notes>SourceType-Scholarly Journals-1</notes><notes>ObjectType-Feature-2</notes><notes>content type line 23</notes><notes>ObjectType-Article-2</notes><notes>ObjectType-Feature-1</notes><abstract>Rett syndrome is a severe neurodevelopmental disorder, with hand stereotypies as a hallmark of the disease. Epilepsy is a frequent comorbidity and is accompanied by centrotemporal spikes on electroencephalogram, but stereotypic movements should not have epileptiform correlates. During routine video-electroencephalographic investigation in 5 Rett syndrome patients, we identified a peculiar type of unilateral, highly rhythmic hand tapping accompanied by contralateral synchronous centrotemporal spikes on electroencephalography. This phenomenon is not consistent with either reflex seizures or hand stereotypies and does not respond to antiepileptic drugs. The electroencephalographic activity probably represents evoked potentials, either somatosensory or motor, whereas the rhythmic activity raises the possibility of a subcortical pacemaker for a stereotypy variant. The phenomenon could be caused by abnormal circuitry among the hyperexcitable somatosensory cortex, motor cortex, and subcortical areas in Rett syndrome. 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subjects | Cerebral Cortex - physiopathology Child Child, Preschool Electroencephalography Evoked Potentials - physiology Female Hand Humans Periodicity Rett Syndrome - physiopathology Stereotyped Behavior - physiology |
title | Unilateral Rhythmic Hand Tapping in Rett Syndrome: Is This Stereotypy? |
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