Dysgraphia as a Mild Expression of Dystonia in Children with Absence Epilepsy
Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia. Based on the clinical observation that children with AE often exhibit, interictally, a disorder resembling writer's cramp but fully definable as dysgraphia, we tested the hypothesis t...
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| description | Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.
Based on the clinical observation that children with AE often exhibit, interictally, a disorder resembling writer's cramp but fully definable as dysgraphia, we tested the hypothesis that in this particular population dysgraphia would represent a subtle expression of dystonia.
We ascertained the prevalence of dysgraphia in 82 children with AE (mean age 9.7) and average intelligence and compared them with 89 age-, gender- and class-matched healthy children (mean age 10.57) using tests for handwriting fluency and quality, based on which we divided patients and controls into four subgroups: AE/dysgraphia, AE without dysgraphia, controls with dysgraphia and healthy controls. We compared the blink reflex recovery cycle in children belonging to all four subgroups.
We identified dysgraphia in 17/82 children with AE and in 7/89 controls (20.7 vs 7.8%; P = 0.016) with the former having a 3.4-times higher risk of dysgraphia regardless of age and gender (odd ratio: 3.49; 95% CI 1.2, 8.8%). The AE/dysgraphia subgroup performed worse than controls with dysgraphia in one test of handwriting fluency (P = 0.037) and in most trials testing handwriting quality (P< 0.02). In children with AE/dysgraphia the blink reflex showed no suppression at short interstimulus intervals, with a difference for each value emerging when comparing the study group with the three remaining subgroups (P |
| doi_str_mv | 10.1371/journal.pone.0130883 |
| format | Article |
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Based on the clinical observation that children with AE often exhibit, interictally, a disorder resembling writer's cramp but fully definable as dysgraphia, we tested the hypothesis that in this particular population dysgraphia would represent a subtle expression of dystonia.
We ascertained the prevalence of dysgraphia in 82 children with AE (mean age 9.7) and average intelligence and compared them with 89 age-, gender- and class-matched healthy children (mean age 10.57) using tests for handwriting fluency and quality, based on which we divided patients and controls into four subgroups: AE/dysgraphia, AE without dysgraphia, controls with dysgraphia and healthy controls. We compared the blink reflex recovery cycle in children belonging to all four subgroups.
We identified dysgraphia in 17/82 children with AE and in 7/89 controls (20.7 vs 7.8%; P = 0.016) with the former having a 3.4-times higher risk of dysgraphia regardless of age and gender (odd ratio: 3.49; 95% CI 1.2, 8.8%). The AE/dysgraphia subgroup performed worse than controls with dysgraphia in one test of handwriting fluency (P = 0.037) and in most trials testing handwriting quality (P< 0.02). In children with AE/dysgraphia the blink reflex showed no suppression at short interstimulus intervals, with a difference for each value emerging when comparing the study group with the three remaining subgroups (P<0.001).
In children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.</description><identifier>ISSN: 1932-6203</identifier><identifier>EISSN: 1932-6203</identifier><identifier>DOI: 10.1371/journal.pone.0130883</identifier><identifier>PMID: 26132164</identifier><language>eng</language><publisher>United States: Public Library of Science</publisher><subject>Adolescent ; Age ; Agraphia - diagnosis ; Agraphia - etiology ; Blink reflex ; Case-Control Studies ; Child ; Children ; Clinical trials ; Cognition & reasoning ; Cognitive ability ; Dysgraphia ; Dystonia ; Dystonia - diagnosis ; Dystonia - etiology ; Epilepsy ; Epilepsy, Absence - complications ; Epilepsy, Absence - diagnosis ; Etiology ; Female ; Handwriting ; Humans ; Informatics ; Intelligence ; Laboratories ; Linguistics ; Male ; Mutation ; Neurology ; Neurosciences ; Pediatrics ; Subgroups ; Substrates ; Writers</subject><ispartof>PloS one, 2015-07, Vol.10 (7), p.e0130883-e0130883</ispartof><rights>COPYRIGHT 2015 Public Library of Science</rights><rights>2015 Guerrini et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>2015 Guerrini et al 2015 Guerrini et al</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c692t-d5409da044d287124db835e66a48a5a288785c4378ff9328225e39302dce38823</citedby><cites>FETCH-LOGICAL-c692t-d5409da044d287124db835e66a48a5a288785c4378ff9328225e39302dce38823</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC4488862/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC4488862/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,860,881,2095,2914,23846,27903,27904,53769,53771,79346,79347</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/26132164$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><contributor>Gonzalez-Alegre, Pedro</contributor><creatorcontrib>Guerrini, Renzo</creatorcontrib><creatorcontrib>Melani, Federico</creatorcontrib><creatorcontrib>Brancati, Claudia</creatorcontrib><creatorcontrib>Ferrari, Anna Rita</creatorcontrib><creatorcontrib>Brovedani, Paola</creatorcontrib><creatorcontrib>Biggeri, Annibale</creatorcontrib><creatorcontrib>Grisotto, Laura</creatorcontrib><creatorcontrib>Pellacani, Simona</creatorcontrib><title>Dysgraphia as a Mild Expression of Dystonia in Children with Absence Epilepsy</title><title>PloS one</title><addtitle>PLoS One</addtitle><description>Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.
Based on the clinical observation that children with AE often exhibit, interictally, a disorder resembling writer's cramp but fully definable as dysgraphia, we tested the hypothesis that in this particular population dysgraphia would represent a subtle expression of dystonia.
We ascertained the prevalence of dysgraphia in 82 children with AE (mean age 9.7) and average intelligence and compared them with 89 age-, gender- and class-matched healthy children (mean age 10.57) using tests for handwriting fluency and quality, based on which we divided patients and controls into four subgroups: AE/dysgraphia, AE without dysgraphia, controls with dysgraphia and healthy controls. We compared the blink reflex recovery cycle in children belonging to all four subgroups.
We identified dysgraphia in 17/82 children with AE and in 7/89 controls (20.7 vs 7.8%; P = 0.016) with the former having a 3.4-times higher risk of dysgraphia regardless of age and gender (odd ratio: 3.49; 95% CI 1.2, 8.8%). The AE/dysgraphia subgroup performed worse than controls with dysgraphia in one test of handwriting fluency (P = 0.037) and in most trials testing handwriting quality (P< 0.02). In children with AE/dysgraphia the blink reflex showed no suppression at short interstimulus intervals, with a difference for each value emerging when comparing the study group with the three remaining subgroups (P<0.001).
In children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.</description><subject>Adolescent</subject><subject>Age</subject><subject>Agraphia - diagnosis</subject><subject>Agraphia - etiology</subject><subject>Blink reflex</subject><subject>Case-Control Studies</subject><subject>Child</subject><subject>Children</subject><subject>Clinical trials</subject><subject>Cognition & reasoning</subject><subject>Cognitive ability</subject><subject>Dysgraphia</subject><subject>Dystonia</subject><subject>Dystonia - diagnosis</subject><subject>Dystonia - etiology</subject><subject>Epilepsy</subject><subject>Epilepsy, Absence - complications</subject><subject>Epilepsy, Absence - diagnosis</subject><subject>Etiology</subject><subject>Female</subject><subject>Handwriting</subject><subject>Humans</subject><subject>Informatics</subject><subject>Intelligence</subject><subject>Laboratories</subject><subject>Linguistics</subject><subject>Male</subject><subject>Mutation</subject><subject>Neurology</subject><subject>Neurosciences</subject><subject>Pediatrics</subject><subject>Subgroups</subject><subject>Substrates</subject><subject>Writers</subject><issn>1932-6203</issn><issn>1932-6203</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2015</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><sourceid>DOA</sourceid><recordid>eNqNkltv0zAYhiMEYgf4BwgiISG4aPEpjnODVJUClTZN4nRrOY6TuHLtzE7Y-u9x12xq0C6QL2zZz_d-B79J8gqCOcQ5_Lhxg7fCzDtn1RxADBjDT5JTWGA0owjgp0fnk-QshA0AGWaUPk9OEIUYQUpOk8vPu9B40bVapCKkIr3UpkpXt51XIWhnU1enEemdjYC26bKN717Z9Eb3bboog7JSpatOG9WF3YvkWS1MUC_H_Tz59WX1c_ltdnH1db1cXMwkLVA_qzICikoAQirEcohIVTKcKUoFYSITiLGcZZLgnNV1bIEhlClcYIAqqTBjCJ8nbw66nXGBj5MIHEb1PCsoYZFYH4jKiQ3vvN4Kv-NOaH534XzDhe-1NIpXQMo6gxXKi4yUJS2EQpKKAmFGAKV11Po0ZhvKrYo12N4LMxGdvljd8sb94YQwxui-3PejgHfXgwo93-oglTHCKjfc1Y1zRHNMIvr2H_Tx7kaqEbEBbWsX88q9KF8QBCAhGSsiNX-EiqtSWy2jber4a9OAD5OAyPTqtm_EEAJf__j-_-zV7yn77ohtlTB9G5wZ-miwMAXJAZTeheBV_TBkCPje9ffT4HvX89H1Mez18Qc9BN3bHP8FV7D59A</recordid><startdate>20150701</startdate><enddate>20150701</enddate><creator>Guerrini, Renzo</creator><creator>Melani, Federico</creator><creator>Brancati, Claudia</creator><creator>Ferrari, Anna Rita</creator><creator>Brovedani, Paola</creator><creator>Biggeri, Annibale</creator><creator>Grisotto, Laura</creator><creator>Pellacani, Simona</creator><general>Public Library of Science</general><general>Public Library of Science (PLoS)</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>IOV</scope><scope>ISR</scope><scope>3V.</scope><scope>7QG</scope><scope>7QL</scope><scope>7QO</scope><scope>7RV</scope><scope>7SN</scope><scope>7SS</scope><scope>7T5</scope><scope>7TG</scope><scope>7TM</scope><scope>7U9</scope><scope>7X2</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8C1</scope><scope>8FD</scope><scope>8FE</scope><scope>8FG</scope><scope>8FH</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABJCF</scope><scope>ABUWG</scope><scope>AEUYN</scope><scope>AFKRA</scope><scope>ARAPS</scope><scope>ATCPS</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BENPR</scope><scope>BGLVJ</scope><scope>BHPHI</scope><scope>C1K</scope><scope>CCPQU</scope><scope>D1I</scope><scope>DWQXO</scope><scope>FR3</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>H94</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>KB.</scope><scope>KB0</scope><scope>KL.</scope><scope>L6V</scope><scope>LK8</scope><scope>M0K</scope><scope>M0S</scope><scope>M1P</scope><scope>M7N</scope><scope>M7P</scope><scope>M7S</scope><scope>NAPCQ</scope><scope>P5Z</scope><scope>P62</scope><scope>P64</scope><scope>PATMY</scope><scope>PDBOC</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>PTHSS</scope><scope>PYCSY</scope><scope>RC3</scope><scope>7X8</scope><scope>5PM</scope><scope>DOA</scope></search><sort><creationdate>20150701</creationdate><title>Dysgraphia as a Mild Expression of Dystonia in Children with Absence Epilepsy</title><author>Guerrini, Renzo ; Melani, Federico ; Brancati, Claudia ; Ferrari, Anna Rita ; Brovedani, Paola ; Biggeri, Annibale ; Grisotto, Laura ; Pellacani, Simona</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c692t-d5409da044d287124db835e66a48a5a288785c4378ff9328225e39302dce38823</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2015</creationdate><topic>Adolescent</topic><topic>Age</topic><topic>Agraphia - 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Academic</collection><collection>PubMed Central (Full Participant titles)</collection><collection>DOAJ Directory of Open Access Journals</collection><jtitle>PloS one</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Guerrini, Renzo</au><au>Melani, Federico</au><au>Brancati, Claudia</au><au>Ferrari, Anna Rita</au><au>Brovedani, Paola</au><au>Biggeri, Annibale</au><au>Grisotto, Laura</au><au>Pellacani, Simona</au><au>Gonzalez-Alegre, Pedro</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Dysgraphia as a Mild Expression of Dystonia in Children with Absence Epilepsy</atitle><jtitle>PloS one</jtitle><addtitle>PLoS One</addtitle><date>2015-07-01</date><risdate>2015</risdate><volume>10</volume><issue>7</issue><spage>e0130883</spage><epage>e0130883</epage><pages>e0130883-e0130883</pages><issn>1932-6203</issn><eissn>1932-6203</eissn><abstract>Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.
Based on the clinical observation that children with AE often exhibit, interictally, a disorder resembling writer's cramp but fully definable as dysgraphia, we tested the hypothesis that in this particular population dysgraphia would represent a subtle expression of dystonia.
We ascertained the prevalence of dysgraphia in 82 children with AE (mean age 9.7) and average intelligence and compared them with 89 age-, gender- and class-matched healthy children (mean age 10.57) using tests for handwriting fluency and quality, based on which we divided patients and controls into four subgroups: AE/dysgraphia, AE without dysgraphia, controls with dysgraphia and healthy controls. We compared the blink reflex recovery cycle in children belonging to all four subgroups.
We identified dysgraphia in 17/82 children with AE and in 7/89 controls (20.7 vs 7.8%; P = 0.016) with the former having a 3.4-times higher risk of dysgraphia regardless of age and gender (odd ratio: 3.49; 95% CI 1.2, 8.8%). The AE/dysgraphia subgroup performed worse than controls with dysgraphia in one test of handwriting fluency (P = 0.037) and in most trials testing handwriting quality (P< 0.02). In children with AE/dysgraphia the blink reflex showed no suppression at short interstimulus intervals, with a difference for each value emerging when comparing the study group with the three remaining subgroups (P<0.001).
In children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>26132164</pmid><doi>10.1371/journal.pone.0130883</doi><oa>free_for_read</oa></addata></record> |
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| subjects | Adolescent Age Agraphia - diagnosis Agraphia - etiology Blink reflex Case-Control Studies Child Children Clinical trials Cognition & reasoning Cognitive ability Dysgraphia Dystonia Dystonia - diagnosis Dystonia - etiology Epilepsy Epilepsy, Absence - complications Epilepsy, Absence - diagnosis Etiology Female Handwriting Humans Informatics Intelligence Laboratories Linguistics Male Mutation Neurology Neurosciences Pediatrics Subgroups Substrates Writers |
| title | Dysgraphia as a Mild Expression of Dystonia in Children with Absence Epilepsy |
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