Literature DB >> 9097959

Iraqi-Jewish kindreds with optic atrophy plus (3-methylglutaconic aciduria type 3) demonstrate linkage disequilibrium with the CTG repeat in the 3' untranslated region of the myotonic dystrophy protein kinase gene.

A Nystuen1, H Costeff, O N Elpeleg, N Apter, B Bonné-Tamir, H Mohrenweiser, N Haider, E M Stone, V C Sheffield.   

Abstract

Iraqi-Jewish optic atrophy plus is an autosomal recessive condition characterized by infantile optic atrophy, an early onset movement disorder, and 3-methylglutaconic aciduria. Other features include spastic paraplegia, mild ataxia, mild cognitive deficiency and dysarthria. This disorder was identified in inbred Iraqi-Jewish kindreds in which relationships between most of the affected individuals were unknown. In this study we identify linkage to chromosome 19q13.2-q13.3 by using a DNA pooling strategy to perform a genome wide screen followed by a high density search for shared segments among affected individuals in candidate regions identified in the initial genome wide screen. A significantly high positive lod score of 6.14 at zero recombination was obtained for the CTG repeat in the 3' untranslated region of the myotonic dystrophy protein kinase gene. The existence of multiple recombinant individuals indicates the disease interval can be further narrowed with additional markers. Linkage disequilibrium was seen in six polymorphic markers across a 1 Mb interval. This region is well characterized and contains several candidate genes.

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Year:  1997        PMID: 9097959     DOI: 10.1093/hmg/6.4.563

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  6 in total

1.  Type III 3-methylglutaconic aciduria (optic atrophy plus syndrome, or Costeff optic atrophy syndrome): identification of the OPA3 gene and its founder mutation in Iraqi Jews.

Authors:  Y Anikster; R Kleta; A Shaag; W A Gahl; O Elpeleg
Journal:  Am J Hum Genet       Date:  2001-10-19       Impact factor: 11.025

2.  OPA3, mutated in 3-methylglutaconic aciduria type III, encodes two transcripts targeted primarily to mitochondria.

Authors:  Marjan Huizing; Heidi Dorward; Lien Ly; Enriko Klootwijk; Robert Kleta; Flemming Skovby; Wuhong Pei; Benjamin Feldman; William A Gahl; Yair Anikster
Journal:  Mol Genet Metab       Date:  2010-03-16       Impact factor: 4.797

Review 3.  Costeff optic atrophy syndrome: new clinical case and novel molecular findings.

Authors:  G Ho; J H Walter; J Christodoulou
Journal:  J Inherit Metab Dis       Date:  2008-11-07       Impact factor: 4.982

4.  Nephrocalcinosis and medullary cysts in 3-methylglutaconic aciduria.

Authors:  Guido F Laube; James V Leonard; William G van't Hoff
Journal:  Pediatr Nephrol       Date:  2003-05-15       Impact factor: 3.714

5.  Costeff syndrome: clinical features and natural history.

Authors:  Gilad Yahalom; Yair Anikster; Ruth Huna-Baron; Chen Hoffmann; Lubov Blumkin; Dorit Lev; Rakefet Tsabari; Zeev Nitsan; Sheera F Lerman; Bruria Ben-Zeev; Ben Pode-Shakked; Shira Sofer; Avraham Schweiger; Tally Lerman-Sagie; Sharon Hassin-Baer
Journal:  J Neurol       Date:  2014-09-09       Impact factor: 4.849

6.  Mitochondrial fusion proteins and human diseases.

Authors:  Michela Ranieri; Simona Brajkovic; Giulietta Riboldi; Dario Ronchi; Federica Rizzo; Nereo Bresolin; Stefania Corti; Giacomo P Comi
Journal:  Neurol Res Int       Date:  2013-05-27
  6 in total

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