Literature DB >> 12545276

Duplication of the MID1 first exon in a patient with Opitz G/BBB syndrome.

Jennifer Winter1, Tanja Lehmann, Vanessa Suckow, Zofia Kijas, Andreas Kulozik, Vera Kalscheuer, Ben Hamel, Koen Devriendt, John Opitz, Steffen Lenzner, Hans-Hilger Ropers, Susann Schweiger.   

Abstract

Opitz G/BBB syndrome is a malformation syndrome of the ventral midline mainly characterized by hypertelorism, swallowing difficulties, hypospadias and developmental delay. SSCP analysis and genomic sequencing of the MID1 open reading frame have identified mutations in 80% of the families with X-linked inheritance. However, in many patients the underlying genetic defect remains undetected by these techniques. Using RNA diagnostics we have now identified a duplication of the MID1 first exon in a patient with X-linked Opitz G/BBB syndrome. This duplication introduces a premature termination codon. In addition, we could significantly lower the threshold for mutation detection on the DNA level by combining SSCP analysis with DHPLC technology.

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Year:  2003        PMID: 12545276     DOI: 10.1007/s00439-002-0901-5

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  12 in total

Review 1.  A perfect message: RNA surveillance and nonsense-mediated decay.

Authors:  M W Hentze; A E Kulozik
Journal:  Cell       Date:  1999-02-05       Impact factor: 41.582

2.  MID1, mutated in Opitz syndrome, encodes an ubiquitin ligase that targets phosphatase 2A for degradation.

Authors:  A Trockenbacher; V Suckow; J Foerster; J Winter; S Krauss; H H Ropers; R Schneider; S Schweiger
Journal:  Nat Genet       Date:  2001-11       Impact factor: 38.330

Review 3.  Distinct phenotypes associated with increasing dosage of the PLP gene: implications for CMT1A due to PMP22 gene duplication.

Authors:  T J Anderson; M Klugmann; C E Thomson; A Schneider; C Readhead; K A Nave; I R Griffiths
Journal:  Ann N Y Acad Sci       Date:  1999-09-14       Impact factor: 5.691

4.  Opitz syndrome is genetically heterogeneous, with one locus on Xp22, and a second locus on 22q11.2.

Authors:  N H Robin; G J Feldman; A L Aronson; H F Mitchell; R Weksberg; C O Leonard; B K Burton; K D Josephson; R Laxová; K A Aleck; J E Allanson; M L Guion-Almeida; R A Martin; L G Leichtman; R A Price; J M Opitz; M Muenke
Journal:  Nat Genet       Date:  1995-12       Impact factor: 38.330

5.  The Opitz syndrome gene product, MID1, associates with microtubules.

Authors:  S Schweiger; J Foerster; T Lehmann; V Suckow; Y A Muller; G Walter; T Davies; H Porter; H van Bokhoven; P W Lunt; P Traub; H H Ropers
Journal:  Proc Natl Acad Sci U S A       Date:  1999-03-16       Impact factor: 11.205

6.  New mutations in MID1 provide support for loss of function as the cause of X-linked Opitz syndrome.

Authors:  T C Cox; L R Allen; L L Cox; B Hopwood; B Goodwin; E Haan; G K Suthers
Journal:  Hum Mol Genet       Date:  2000-10-12       Impact factor: 6.150

7.  Characterization and physical mapping in human and mouse of a novel RING finger gene in Xp22.

Authors:  I B Van den Veyver; T A Cormier; V Jurecic; A Baldini; H Y Zoghbi
Journal:  Genomics       Date:  1998-07-15       Impact factor: 5.736

8.  Alu-Alu recombination results in a duplication of seven exons in the lysyl hydroxylase gene in a patient with the type VI variant of Ehlers-Danlos syndrome.

Authors:  B Pousi; T Hautala; J Heikkinen; L Pajunen; K I Kivirikko; R Myllylä
Journal:  Am J Hum Genet       Date:  1994-11       Impact factor: 11.025

9.  Opitz G/BBB syndrome in Xp22: mutations in the MID1 gene cluster in the carboxy-terminal domain.

Authors:  K Gaudenz; E Roessler; N Quaderi; B Franco; G Feldman; D L Gasser; B Wittwer; J Horst; E Montini; J M Opitz; A Ballabio; M Muenke
Journal:  Am J Hum Genet       Date:  1998-09       Impact factor: 11.025

10.  Opitz G/BBB syndrome, a defect of midline development, is due to mutations in a new RING finger gene on Xp22.

Authors:  N A Quaderi; S Schweiger; K Gaudenz; B Franco; E I Rugarli; W Berger; G J Feldman; M Volta; G Andolfi; S Gilgenkrantz; R W Marion; R C Hennekam; J M Opitz; M Muenke; H H Ropers; A Ballabio
Journal:  Nat Genet       Date:  1997-11       Impact factor: 38.330

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  12 in total

1.  Regulation of the MID1 protein function is fine-tuned by a complex pattern of alternative splicing.

Authors:  Jennifer Winter; Tanja Lehmann; Sybille Krauss; Alexander Trockenbacher; Zofia Kijas; John Foerster; Vanessa Suckow; Marie-Laure Yaspo; Andreas Kulozik; Vera Kalscheuer; Rainer Schneider; Susann Schweiger
Journal:  Hum Genet       Date:  2004-03-31       Impact factor: 4.132

2.  aura (mid1ip1l) regulates the cytoskeleton at the zebrafish egg-to-embryo transition.

Authors:  Celeste Eno; Bharti Solanki; Francisco Pelegri
Journal:  Development       Date:  2016-03-10       Impact factor: 6.868

3.  Genetic heterogeneity in Rubinstein-Taybi syndrome: mutations in both the CBP and EP300 genes cause disease.

Authors:  Jeroen H Roelfsema; Stefan J White; Yavuz Ariyürek; Deborah Bartholdi; Dunja Niedrist; Francesco Papadia; Carlos A Bacino; Johan T den Dunnen; Gert-Jan B van Ommen; Martijn H Breuning; Raoul C Hennekam; Dorien J M Peters
Journal:  Am J Hum Genet       Date:  2005-02-10       Impact factor: 11.025

4.  Modulation of F-actin dynamics by maternal Mid1ip1L controls germ plasm aggregation and furrow recruitment in the zebrafish embryo.

Authors:  Celeste Eno; Francisco Pelegri
Journal:  Development       Date:  2018-05-17       Impact factor: 6.868

5.  Two Novel Pathogenic MID1 Variants and Genotype-Phenotype Correlation Reanalysis in X-Linked Opitz G/BBB Syndrome.

Authors:  Nuno Maia; Maria J Nabais Sá; Nataliya Tkachenko; Gabriela Soares; Isabel Marques; Bárbara Rodrigues; Ana M Fortuna; Rosário Santos; Arjan P M de Brouwer; Paula Jorge
Journal:  Mol Syndromol       Date:  2017-08-29

6.  Mig12, a novel Opitz syndrome gene product partner, is expressed in the embryonic ventral midline and co-operates with Mid1 to bundle and stabilize microtubules.

Authors:  Caterina Berti; Bianca Fontanella; Rosa Ferrentino; Germana Meroni
Journal:  BMC Cell Biol       Date:  2004-02-29       Impact factor: 4.241

7.  Molecular dynamics simulation reveals insights into the mechanism of unfolding by the A130T/V mutations within the MID1 zinc-binding Bbox1 domain.

Authors:  Yunjie Zhao; Chen Zeng; Michael A Massiah
Journal:  PLoS One       Date:  2015-04-13       Impact factor: 3.240

8.  The Challenge of Prenatal Diagnostic Work-Up of Maternally Inherited X-Linked Opitz G/BBB: Case Report and Literature Review.

Authors:  Marialuigia Spinelli; Carmine Sica; Bruno Dallapiccola; Antonio Novelli; Letizia Di Meglio; Pasquale Martinelli
Journal:  Case Rep Obstet Gynecol       Date:  2015-05-04

9.  Alternative polyadenylation signals and promoters act in concert to control tissue-specific expression of the Opitz Syndrome gene MID1.

Authors:  Jennifer Winter; Melanie Kunath; Stefan Roepcke; Sven Krause; Rainer Schneider; Susann Schweiger
Journal:  BMC Mol Biol       Date:  2007-11-15       Impact factor: 2.946

10.  XLOS-observed mutations of MID1 Bbox1 domain cause domain unfolding.

Authors:  Katharine M Wright; Kuanlin Wu; Omotolani Babatunde; Haijuan Du; Michael A Massiah
Journal:  PLoS One       Date:  2014-09-12       Impact factor: 3.240

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