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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-12-38-a</article-id>
<article-id pub-id-type="doi">10.15844/pedneurbriefs-12-5-9</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Heredo-Degenerative 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>Clinical and Genetic Diagnosis of Friedreich&#x2019;s Ataxia</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>05</month>
<year>1998</year>
</pub-date>
<pub-date date-type="pub" publication-format="electronic">
<day>01</day>
<month>05</month>
<year>2016</year>
</pub-date>
<volume>12</volume>
<issue>5</issue>
<fpage>38</fpage>
<lpage>38</lpage>
<permissions>
<copyright-statement>Copyright: &#x00A9; 1998 The Author(s)</copyright-statement>
<copyright-year>1998</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="10.1136/adc.78.3.204" vol="78" page="204">
<article-title>Diagnosing Friedreich's ataxia</article-title>
</related-article>
<abstract abstract-type="web-summary" specific-use="electronic-only">
<p>The clinical diagnostic criteria and genetic testing for Friedreich&#x2019;s ataxia are reviewed from the National Hospital for Neurology and Neurosurgery, Queen Square, London, UK.</p>
</abstract>
<kwd-group>
<kwd>Clinical Diagnostic Criteria</kwd>
<kwd>Progressive Ataxia</kwd>
<kwd>Cardiomyopathy</kwd>
</kwd-group>
</article-meta>
</front>
<body>
<p>The clinical diagnostic criteria and genetic testing for Friedreich&#x2019;s ataxia are reviewed from the National Hospital for Neurology and Neurosurgery, Queen Square, London, UK. The essential clinical diagnostic criteria, after Harding, are onset before 25 years, progressive ataxia, absent tendon reflexes, axonopathy, and dysarthria. Additional criteria include scoliosis, cardiomyopathy, optic atrophy, pes cavus, and diabetes. The gene for Friedreich&#x2019;s ataxia is mapped to chromosome 9q13, with an X25 transcript and GAA mutation repeat in intron 1 of the frataxin gene. The repeat length is correlated with the age at onset and the presence of cardiomyopathy. The frataxin protein may be an iron transporter within the mitochondria. [<xref ref-type="bibr" rid="CIT0001">1</xref>]</p>
<p>COMMENT. A direct genetic test permits diagnosis of Friedreich&#x2019;s ataxia in forme fruste cases, including those with retained reflexes or onset later than 25 years. The author postulates that the clinical manifestations of Friedreich&#x2019;s ataxia coupled with the nature of the frataxin protein have the hallmarks of a mitochondrial disease.</p>
</body>
<back>
<ref-list>
<ref id="CIT0001">
<label>1</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name>
<surname>Wood</surname>
<given-names>NW</given-names>
</name>
</person-group>
<article-title>Diagnosing Friedreich's ataxia</article-title>
<source>Arch Dis Child</source>
<year>1998</year>
<month>Mar</month>
<volume>78</volume>
<issue>3</issue>
<fpage>204</fpage>
<lpage>7</lpage>
<pub-id pub-id-type="doi">10.1136/adc.78.3.204</pub-id>
<pub-id pub-id-type="pmid">9613347</pub-id>
</element-citation>
</ref>
</ref-list>
</back>
</article>
