• Media type: E-Article
  • Title: A Novel Mutation in the ROGDI Gene in a Patient with Kohlschütter-Tönz Syndrome
  • Contributor: Huckert, Mathilde; Mecili, Helen; Laugel-Haushalter, Virginie; Stoetzel, Corinne; Muller, Jean; Flori, Elisabeth; Laugel, Vincent; Manière, Marie-Cécile; Dollfus, Hélène; Bloch-Zupan, Agnès
  • imprint: S. Karger AG, 2014
  • Published in: Molecular Syndromology
  • Language: English
  • DOI: 10.1159/000366252
  • ISSN: 1661-8769; 1661-8777
  • Keywords: Genetics (clinical) ; Genetics
  • Origination:
  • Footnote:
  • Description: <jats:p>Kohlschütter-Tönz Syndrome (KTZS) is an autosomal recessive disorder caused by mutations in the &lt;i&gt;ROGDI&lt;/i&gt; gene. This syndrome is characterized by epilepsy, psychomotor regression and amelogenesis imperfecta. In this paper, we report a case of a 13-year-old Malian girl presenting with this rare disease. By genetic analysis, we identified a novel&lt;i&gt; ROGDI&lt;/i&gt; homozygous mutation NM_024589.1: c.117+1G&gt;T [Chr16 (GRCh37): g.4852382C&gt;A] which confirmed the diagnosis of Kohlschütter-Tönz syndrome. The mutation abolishes the usual splice donor site of intron 2 which leads to the deletion of exon 2 and in-frame assembly of exon 3. Exon 2 encodes a highly conserved leucine-rich region that is essential for ROGDI protein function. Hence, this deletion may affect the function of the ROGDI protein.</jats:p>
  • Access State: Open Access