Neuromotor deficits in children with the 22q11 deletion syndrome.
Identifieur interne : 002A77 ( PubMed/Corpus ); précédent : 002A76; suivant : 002A78Neuromotor deficits in children with the 22q11 deletion syndrome.
Auteurs : Christina Sobin ; Samantha H. Monk ; Karen Kiley-Brabeck ; Jananne Khuri ; Maria KarayiorgouSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2006.
English descriptors
- KwdEn :
- Adolescent, Age Factors, Age of Onset, Chi-Square Distribution, Child, Child, Preschool, Chromosome Deletion, Chromosome Disorders (complications), Chromosome Disorders (genetics), Chromosomes, Human, Pair 22, Developmental Disabilities, Disease Progression, Female, Humans, Intelligence (physiology), Longitudinal Studies, Male, Movement Disorders (etiology), Movement Disorders (genetics), Sex Factors, Siblings.
- MESH :
- complications : Chromosome Disorders.
- etiology : Movement Disorders.
- genetics : Chromosome Disorders, Movement Disorders.
- physiology : Intelligence.
- Adolescent, Age Factors, Age of Onset, Chi-Square Distribution, Child, Child, Preschool, Chromosome Deletion, Chromosomes, Human, Pair 22, Developmental Disabilities, Disease Progression, Female, Humans, Longitudinal Studies, Male, Sex Factors, Siblings.
Abstract
The 22q11 chromosomal deletion syndrome (22q11DS) is associated with a heterogeneous physical phenotype, neurocognitive deficits, and increased risk of later psychiatric illness. Sporadic clinical reports suggested motor differences, but quantitative studies of movement in children with 22q11DS are rare. If present in a majority of affected school-age children, characterization of neuromotor deficits may prove to be critical for intervention, neurocognitive test interpretation, and understanding etiology. We administered the Movement Assessment Battery for Children to 72 children ages 4.3 to 16.1, including 49 children confirmed positive for the 22q11 deletion and 23 control siblings. We predicted a higher frequency of global and domain impairment in manual dexterity, eye-hand coordination, and balance among affected children. Ninety-four percent of affected children had marked neuromotor deficits, and group scores differed broadly for both global and subarea measures. Secondary analyses showed no impairment differences between younger and older children with 22q11DS, and longitudinal trajectories for 12 affected children suggested stability of deficits over 3-year intervals. Neuromotor deficits in children with 22q11DS occur early in development, continue throughout the school-age years, should be considered in the interpretation of motor-based achievement and IQ tests, and require targeted and ongoing remediation throughout childhood and adolescence. Further studies examining the specificity of motor impairment to 22q11DS are needed.
DOI: 10.1002/mds.21103
PubMed: 16991148
Links to Exploration step
pubmed:16991148Le document en format XML
<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Neuromotor deficits in children with the 22q11 deletion syndrome.</title><author><name sortKey="Sobin, Christina" sort="Sobin, Christina" uniqKey="Sobin C" first="Christina" last="Sobin">Christina Sobin</name><affiliation><nlm:affiliation>Rockefeller University, New York, New York, USA. casobin@utep.edu</nlm:affiliation></affiliation></author><author><name sortKey="Monk, Samantha H" sort="Monk, Samantha H" uniqKey="Monk S" first="Samantha H" last="Monk">Samantha H. Monk</name></author><author><name sortKey="Kiley Brabeck, Karen" sort="Kiley Brabeck, Karen" uniqKey="Kiley Brabeck K" first="Karen" last="Kiley-Brabeck">Karen Kiley-Brabeck</name></author><author><name sortKey="Khuri, Jananne" sort="Khuri, Jananne" uniqKey="Khuri J" first="Jananne" last="Khuri">Jananne Khuri</name></author><author><name sortKey="Karayiorgou, Maria" sort="Karayiorgou, Maria" uniqKey="Karayiorgou M" first="Maria" last="Karayiorgou">Maria Karayiorgou</name></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2006">2006</date><idno type="doi">10.1002/mds.21103</idno><idno type="RBID">pubmed:16991148</idno><idno type="pmid">16991148</idno><idno type="wicri:Area/PubMed/Corpus">002A77</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Neuromotor deficits in children with the 22q11 deletion syndrome.</title><author><name sortKey="Sobin, Christina" sort="Sobin, Christina" uniqKey="Sobin C" first="Christina" last="Sobin">Christina Sobin</name><affiliation><nlm:affiliation>Rockefeller University, New York, New York, USA. casobin@utep.edu</nlm:affiliation></affiliation></author><author><name sortKey="Monk, Samantha H" sort="Monk, Samantha H" uniqKey="Monk S" first="Samantha H" last="Monk">Samantha H. Monk</name></author><author><name sortKey="Kiley Brabeck, Karen" sort="Kiley Brabeck, Karen" uniqKey="Kiley Brabeck K" first="Karen" last="Kiley-Brabeck">Karen Kiley-Brabeck</name></author><author><name sortKey="Khuri, Jananne" sort="Khuri, Jananne" uniqKey="Khuri J" first="Jananne" last="Khuri">Jananne Khuri</name></author><author><name sortKey="Karayiorgou, Maria" sort="Karayiorgou, Maria" uniqKey="Karayiorgou M" first="Maria" last="Karayiorgou">Maria Karayiorgou</name></author></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="ISSN">0885-3185</idno><imprint><date when="2006" type="published">2006</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent</term><term>Age Factors</term><term>Age of Onset</term><term>Chi-Square Distribution</term><term>Child</term><term>Child, Preschool</term><term>Chromosome Deletion</term><term>Chromosome Disorders (complications)</term><term>Chromosome Disorders (genetics)</term><term>Chromosomes, Human, Pair 22</term><term>Developmental Disabilities</term><term>Disease Progression</term><term>Female</term><term>Humans</term><term>Intelligence (physiology)</term><term>Longitudinal Studies</term><term>Male</term><term>Movement Disorders (etiology)</term><term>Movement Disorders (genetics)</term><term>Sex Factors</term><term>Siblings</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Chromosome Disorders</term></keywords><keywords scheme="MESH" qualifier="etiology" xml:lang="en"><term>Movement Disorders</term></keywords><keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Chromosome Disorders</term><term>Movement Disorders</term></keywords><keywords scheme="MESH" qualifier="physiology" xml:lang="en"><term>Intelligence</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Age Factors</term><term>Age of Onset</term><term>Chi-Square Distribution</term><term>Child</term><term>Child, Preschool</term><term>Chromosome Deletion</term><term>Chromosomes, Human, Pair 22</term><term>Developmental Disabilities</term><term>Disease Progression</term><term>Female</term><term>Humans</term><term>Longitudinal Studies</term><term>Male</term><term>Sex Factors</term><term>Siblings</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">The 22q11 chromosomal deletion syndrome (22q11DS) is associated with a heterogeneous physical phenotype, neurocognitive deficits, and increased risk of later psychiatric illness. Sporadic clinical reports suggested motor differences, but quantitative studies of movement in children with 22q11DS are rare. If present in a majority of affected school-age children, characterization of neuromotor deficits may prove to be critical for intervention, neurocognitive test interpretation, and understanding etiology. We administered the Movement Assessment Battery for Children to 72 children ages 4.3 to 16.1, including 49 children confirmed positive for the 22q11 deletion and 23 control siblings. We predicted a higher frequency of global and domain impairment in manual dexterity, eye-hand coordination, and balance among affected children. Ninety-four percent of affected children had marked neuromotor deficits, and group scores differed broadly for both global and subarea measures. Secondary analyses showed no impairment differences between younger and older children with 22q11DS, and longitudinal trajectories for 12 affected children suggested stability of deficits over 3-year intervals. Neuromotor deficits in children with 22q11DS occur early in development, continue throughout the school-age years, should be considered in the interpretation of motor-based achievement and IQ tests, and require targeted and ongoing remediation throughout childhood and adolescence. Further studies examining the specificity of motor impairment to 22q11DS are needed.</div></front></TEI><pubmed><MedlineCitation Owner="NLM" Status="MEDLINE"><PMID Version="1">16991148</PMID><DateCreated><Year>2006</Year><Month>12</Month><Day>27</Day></DateCreated><DateCompleted><Year>2007</Year><Month>02</Month><Day>20</Day></DateCompleted><DateRevised><Year>2014</Year><Month>09</Month><Day>08</Day></DateRevised><Article PubModel="Print"><Journal><ISSN IssnType="Print">0885-3185</ISSN><JournalIssue CitedMedium="Print"><Volume>21</Volume><Issue>12</Issue><PubDate><Year>2006</Year><Month>Dec</Month></PubDate></JournalIssue><Title>Movement disorders : official journal of the Movement Disorder Society</Title><ISOAbbreviation>Mov. Disord.</ISOAbbreviation></Journal><ArticleTitle>Neuromotor deficits in children with the 22q11 deletion syndrome.</ArticleTitle><Pagination><MedlinePgn>2082-9</MedlinePgn></Pagination><Abstract><AbstractText>The 22q11 chromosomal deletion syndrome (22q11DS) is associated with a heterogeneous physical phenotype, neurocognitive deficits, and increased risk of later psychiatric illness. Sporadic clinical reports suggested motor differences, but quantitative studies of movement in children with 22q11DS are rare. If present in a majority of affected school-age children, characterization of neuromotor deficits may prove to be critical for intervention, neurocognitive test interpretation, and understanding etiology. We administered the Movement Assessment Battery for Children to 72 children ages 4.3 to 16.1, including 49 children confirmed positive for the 22q11 deletion and 23 control siblings. We predicted a higher frequency of global and domain impairment in manual dexterity, eye-hand coordination, and balance among affected children. Ninety-four percent of affected children had marked neuromotor deficits, and group scores differed broadly for both global and subarea measures. Secondary analyses showed no impairment differences between younger and older children with 22q11DS, and longitudinal trajectories for 12 affected children suggested stability of deficits over 3-year intervals. Neuromotor deficits in children with 22q11DS occur early in development, continue throughout the school-age years, should be considered in the interpretation of motor-based achievement and IQ tests, and require targeted and ongoing remediation throughout childhood and adolescence. Further studies examining the specificity of motor impairment to 22q11DS are needed.</AbstractText><CopyrightInformation>Copyright 2006 Movement Disorder Society.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Sobin</LastName><ForeName>Christina</ForeName><Initials>C</Initials><AffiliationInfo><Affiliation>Rockefeller University, New York, New York, USA. casobin@utep.edu</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Monk</LastName><ForeName>Samantha H</ForeName><Initials>SH</Initials></Author><Author ValidYN="Y"><LastName>Kiley-Brabeck</LastName><ForeName>Karen</ForeName><Initials>K</Initials></Author><Author ValidYN="Y"><LastName>Khuri</LastName><ForeName>Jananne</ForeName><Initials>J</Initials></Author><Author ValidYN="Y"><LastName>Karayiorgou</LastName><ForeName>Maria</ForeName><Initials>M</Initials></Author></AuthorList><Language>eng</Language><GrantList CompleteYN="Y"><Grant><GrantID>K08 HD040321</GrantID><Acronym>HD</Acronym><Agency>NICHD NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>K08 HD040321-05</GrantID><Acronym>HD</Acronym><Agency>NICHD NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>K08-HD040321</GrantID><Acronym>HD</Acronym><Agency>NICHD NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>M01-RR00102</GrantID><Acronym>RR</Acronym><Agency>NCRR NIH HHS</Agency><Country>United States</Country></Grant></GrantList><PublicationTypeList><PublicationType UI="D003160">Comparative Study</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D052061">Research Support, N.I.H., Extramural</PublicationType></PublicationTypeList></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Mov Disord</MedlineTA><NlmUniqueID>8610688</NlmUniqueID><ISSNLinking>0885-3185</ISSNLinking></MedlineJournalInfo><CitationSubset>IM</CitationSubset><CommentsCorrectionsList><CommentsCorrections RefType="Cites"><RefSource>Lakartidningen. 1999 Nov 3;96(44):4789-93</RefSource><PMID Version="1">10584540</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Dev Med Child Neurol. 2000 Jun;42(6):422-7</RefSource><PMID Version="1">10875531</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Child Neuropsychol. 1999 Dec;5(4):230-41</RefSource><PMID Version="1">10925707</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Percept Mot Skills. 2001 Aug;93(1):275-80</RefSource><PMID Version="1">11693695</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>J Clin Exp Neuropsychol. 2001 Aug;23(4):447-64</RefSource><PMID Version="1">11780945</PMID></CommentsCorrections><CommentsCorrections 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