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<front>
<journal-meta>
<journal-id journal-id-type="issn">1043-3155</journal-id>
<journal-id journal-id-type="nlm-ta">Pediatr Neurol Briefs</journal-id>
<journal-id journal-id-type="pmc">pedneurbriefs</journal-id>
<journal-id journal-id-type="iso-abbrev">Pediatr Neurol Briefs</journal-id>
<journal-title-group>
<journal-title>Pediatric Neurology Briefs</journal-title>
<abbrev-journal-title>Pediatr Neurol Briefs</abbrev-journal-title>
</journal-title-group>
<issn pub-type="epub">2166-6482</issn>
<issn pub-type="ppub">1043-3155</issn>
<issn-l>2166-3155</issn-l>
<publisher>
<publisher-name>Pediatric Neurology Briefs Publishers</publisher-name>
<publisher-loc>Chicago, IL, USA</publisher-loc>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="publisher-id">PNB-22-26</article-id>
<article-id pub-id-type="doi">10.15844/pedneurbriefs-22-4-2</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Seizure Disorders</subject>
</subj-group>
<subj-group subj-group-type="Discipline-v2">
<subject>Neurology</subject>
<subject>Pediatrics</subject>
<subject>Nervous System Diseases</subject>
<subject>Child Development</subject>
<subject>Brain Diseases</subject>
<subject>Neurosurgery</subject>
<subject>Child</subject>
<subject>Infant</subject>
</subj-group>
</article-categories>
<title-group>
<article-title>Genetics of Febrile Seizures and Epilepsy (GEFS+)</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author" corresp="yes">
<contrib-id contrib-id-type="orcid">http://orcid.org/0000-0002-0173-7931</contrib-id>
<name>
<surname>Millichap</surname>
<given-names>J. Gordon</given-names>
</name>
<degrees>MD</degrees>
<xref ref-type="aff" rid="AF0001">1</xref>
<xref ref-type="aff" rid="AF0002">2</xref>
<xref ref-type="corresp" rid="cor1">&#x002A;</xref>
</contrib>
</contrib-group>
<aff id="AF0001">
<label>1</label>Division of Neurology, Children&#x0027;s Memorial Hospital, Chicago, IL</aff>
<aff id="AF0002">
<label>2</label>Departments of Pediatrics and Neurology, Northwestern University Feinberg School of Medicine, Chicago, IL</aff>
<author-notes>
<corresp id="cor1"><label>&#x002A;</label>Correspondence: Dr. J. Gordon Millichap, E-mail: <email xlink:href="jgmillichap@northwestern.edu">jgmillichap@northwestern.edu</email>
</corresp>
</author-notes>
<pub-date date-type="pub" publication-format="print">
<month>04</month>
<year>2008</year>
</pub-date>
<pub-date date-type="pub" publication-format="electronic">
<day>01</day>
<month>02</month>
<year>2016</year>
</pub-date>
<volume>22</volume>
<issue>4</issue>
<fpage>26</fpage>
<lpage>27</lpage>
<permissions>
<copyright-statement>Copyright: &#x00A9; 2008 The Author(s)</copyright-statement>
<copyright-year>2008</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/4.0/">
<license-p>This work is licensed under the <uri xlink:href="http://creativecommons.org/licenses/by/4.0/">Creative Commons Attribution 4.0 International License</uri>, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
</license>
</permissions>
<related-article id="R1" related-article-type="commentary-article" ext-link-type="doi" xlink:href="10.1111/j.1600-0404.2007.00941.x" vol="117" page="289">
<article-title>Genetic screening of Scandinavian families with febrile seizures and epilepsy or GEFS+</article-title>
</related-article>
<abstract abstract-type="web-summary" specific-use="electronic-only">
<p>Mutations in 3 genes <italic>SCN1A, SCN1B</italic> and <italic>GABRG2</italic> have been shown to cause GEFS+ in families of various ethnic origins.</p>
</abstract>
<kwd-group>
<kwd>SCN1A</kwd>
<kwd>Autosomal Dominant Disorder</kwd>
<kwd>G-Aminobutyric Acid</kwd>
</kwd-group>
</article-meta>
</front>
<body>
<p>Mutations in 3 genes <italic>SCN1A, SCN1B</italic> and <italic>GABRG2</italic> have been shown to cause GEFS+ in families of various ethnic origins. The occurrence of mutations in these genes in 19 families of Scandinavian origin with a history of GEFS+ was studied at Ulleval University Hospital, Oslo, Norway, and centers in Denmark. Families were identified from population-based twin registries in Denmark and Norway. One mutation in <italic>SCN1A</italic> was identified in a Danish family with phenotypes consistent with GEFS+. The mutation was not found in healthy and unrelated controls. No mutations were found in any of the other families. [<xref ref-type="bibr" rid="CIT0001">1</xref>]</p>
<p>COMMENT. GEFS+ is an autosomal dominant disorder characterized by multiple febrile seizures persisting beyond age 5 years and complicated by afebrile seizures of absence, myoclonic or atonic types. Seizures cease in mid-childhood [<xref ref-type="bibr" rid="CIT0002">2</xref>, <xref ref-type="bibr" rid="CIT0003">3</xref>]. Genes on chromosomes 2q24 and 19ql3 encode subunits of the voltage-gated sodium ion channels, while the gene on 5q31 codes for the g-subunit of the g-aminobutyric acid (GABA) receptor. The genes responsible for GEFS+ show considerable heterogeneity and variable expressivity. GEFS+ is an evolving composite of many syndromes, with shared genetic susceptibility [<xref ref-type="bibr" rid="CIT0004">4</xref>]. While the definition of GEFS+ is continually changing and probably involves many genes, the common denominator is the association with febrile seizures.</p>
<p><bold>Failure of replication of epilepsy gene associations</bold> is discussed by researchers from Columbia University Medical Center, and New York State Psychiatric Institute, New York, NY (<xref ref-type="bibr" rid="CIT0005">5</xref>). Over 50 genetic associations with various idiopathic epilepsy syndromes are reported but most have not been replicated. Genetic heterogeneity is a confounder in population-based studies, in both association and linkage studies. Linkage, association, and mutation analyses are the most common methods of evaluating candidate genes in epilepsy. The authors advocate the integration of results from different experimental methods rather than insisting only on replication.</p>
<p>Discovery of susceptibility genes and their association with drug responsiveness and sideeffects should permit new diagnostic and therapeutic options in the management of the epilepsies. [<xref ref-type="bibr" rid="CIT0006">6</xref>]</p>
</body>
<back>
<ref-list>
<ref id="CIT0001">
<label>1</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name>
<surname>Selmer</surname>
<given-names>KK</given-names>
</name>
<name>
<surname>Egeland</surname>
<given-names>T</given-names>
</name>
<name>
<surname>Solaas</surname>
<given-names>MH</given-names>
</name>
<name>
<surname>Nakken</surname>
<given-names>KO</given-names>
</name>
<name>
<surname>Kjeldsen</surname>
<given-names>MJ</given-names>
</name>
<name>
<surname>Friis</surname>
<given-names>ML</given-names>
</name>
<etal/>
</person-group>
<article-title>Genetic screening of Scandinavian families with febrile seizures and epilepsy or GEFS+</article-title>
<source>Acta Neurol Scand</source>
<year>2008</year>
<month>April</month>
<volume>117</volume>
<issue>4</issue>
<fpage>289</fpage>
<lpage>292</lpage>
<pub-id pub-id-type="doi">10.1111/j.1600-0404.2007.00941.x</pub-id>
<pub-id pub-id-type="pmid">17927801</pub-id>
</element-citation>
</ref>
<ref id="CIT0002">
<label>2</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name>
<surname>Scheffer</surname>
<given-names>IE</given-names>
</name>
<name>
<surname>Berkovic</surname>
<given-names>SF</given-names>
</name>
</person-group>
<article-title>Generalized epilepsy with febrile seizures plus. A genetic disorder with heterogeneous clinical phenotypes</article-title>
<source>Brain</source>
<year>1997</year>
<month>Mar</month>
<volume>120</volume>
<issue>Pt 3</issue>
<fpage>479</fpage>
<lpage>490</lpage>
<pub-id pub-id-type="doi">10.1093/brain/120.3.479</pub-id>
<pub-id pub-id-type="pmid">9126059</pub-id>
</element-citation>
</ref>
<ref id="CIT0003">
<label>3</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name>
<surname>Scheffer</surname>
<given-names>IE</given-names>
</name>
<name>
<surname>Harkin</surname>
<given-names>LA</given-names>
</name>
<name>
<surname>Dibbens</surname>
<given-names>LM</given-names>
</name>
<name>
<surname>Mulley</surname>
<given-names>JC</given-names>
</name>
<name>
<surname>Berkovic</surname>
<given-names>SF</given-names>
</name>
</person-group>
<article-title>Neonatal epilepsy syndromes and generalized epilepsy with febrile seizures plus (GEFS+)</article-title>
<source>Epilepsia</source>
<year>2005</year>
<volume>46</volume>
<supplement>Suppl 10</supplement>
<fpage>41</fpage>
<lpage>47</lpage>
<pub-id pub-id-type="doi">10.1111/j.1528-1167.2005.00358.x</pub-id>
<pub-id pub-id-type="pmid">16359471</pub-id>
</element-citation>
</ref>
<ref id="CIT0004">
<label>4</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name>
<surname>Nordli</surname>
<given-names>DR</given-names>
<suffix>Jr</suffix>
</name>
</person-group>
<article-title>Idiopathic generalized epilepsies recognized by the International League Against Epilepsy</article-title>
<source>Epilepsia</source>
<year>2005</year>
<volume>46</volume>
<supplement>Suppl 9</supplement>
<fpage>48</fpage>
<lpage>56</lpage>
<pub-id pub-id-type="doi">10.1111/j.1528-1167.2005.00313.x</pub-id>
<pub-id pub-id-type="pmid">16302875</pub-id>
</element-citation>
</ref>
<ref id="CIT0005">
<label>5</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name>
<surname>Pal</surname>
<given-names>DK</given-names>
</name>
<name>
<surname>Strug</surname>
<given-names>LJ</given-names>
</name>
<name>
<surname>Greenberg</surname>
<given-names>DA</given-names>
</name>
</person-group>
<article-title>Evaluating candidate genes in common epilepsies and the nature of evidence</article-title>
<source>Epilepsia</source>
<year>2008</year>
<month>Mar</month>
<volume>49</volume>
<issue>3</issue>
<fpage>386</fpage>
<lpage>392</lpage>
<pub-id pub-id-type="doi">10.1111/j.1528-1167.2007.01416.x</pub-id>
<pub-id pub-id-type="pmid">18028406</pub-id>
</element-citation>
</ref>
<ref id="CIT0006">
<label>6</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name>
<surname>Helbig</surname>
<given-names>I</given-names>
</name>
<name>
<surname>Scheffer</surname>
<given-names>IE</given-names>
</name>
<name>
<surname>Mulley</surname>
<given-names>JC</given-names>
</name>
<name>
<surname>Berkovic</surname>
<given-names>SF</given-names>
</name>
</person-group>
<article-title>Navigating the channels and beyond: unravelling the genetics of the epilepsies</article-title>
<source>Lancet Neurol</source>
<year>2008</year>
<month>March</month>
<volume>7</volume>
<issue>3</issue>
<fpage>231</fpage>
<lpage>245</lpage>
<pub-id pub-id-type="doi">10.1016/S1474-4422(08)70039-5</pub-id>
<pub-id pub-id-type="pmid">18275925</pub-id>
</element-citation>
</ref>
</ref-list>
</back>
</article>
