Literature DB >> 8268910

Isolation of a zinc finger gene consistently deleted in DiGeorge syndrome.

M Aubry1, S Demczuk, C Desmaze, M Aikem, A Aurias, J P Julien, G A Rouleau.   

Abstract

DiGeorge syndrome is a human developmental disorder resulting in hypoplasia of the thymus and parathyroids, and conotruncal heart defects. We recently isolated four genes with zinc finger DNA binding motifs mapping to chromosome 22q11.2 DiGeorge critical region. We now report that one of them, ZNF74 gene, is hemizygously deleted in 23 out of 24 DiGeorge syndrome patients tested. ZNF74 mRNA transcripts are detected in human and mouse embryos but not in adult tissues. Sequence analysis of a corresponding cDNA reveals an an open reading frame encoding 12 zinc finger motifs of the Kruppel/TFIIIA type as well as N-terminal and C-terminal non-zinc finger domains. These results suggest that changes in the dosage of a putative transcription factor through ZNF74 hemizygous deletion may be critical for DiGeorge developmental anomalies.

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Year:  1993        PMID: 8268910     DOI: 10.1093/hmg/2.10.1583

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


  8 in total

1.  An unusual concurrence of graft versus host disease caused by engraftment of maternal lymphocytes with DiGeorge anomaly.

Authors:  J G Ocejo-Vinyals; M J Lozano; P Sánchez-Velasco; J Escribano de Diego; J E Paz-Miguel; F Leyva-Cobián
Journal:  Arch Dis Child       Date:  2000-08       Impact factor: 3.791

2.  Der(22) syndrome and velo-cardio-facial syndrome/DiGeorge syndrome share a 1.5-Mb region of overlap on chromosome 22q11.

Authors:  B Funke; L Edelmann; N McCain; R K Pandita; J Ferreira; S Merscher; M Zohouri; L Cannizzaro; A Shanske; B E Morrow
Journal:  Am J Hum Genet       Date:  1999-03       Impact factor: 11.025

3.  KRAB-independent suppression of neoplastic cell growth by the novel zinc finger transcription factor KS1.

Authors:  B Gebelein; M Fernandez-Zapico; M Imoto; R Urrutia
Journal:  J Clin Invest       Date:  1998-12-01       Impact factor: 14.808

4.  Juvenile rheumatoid arthritis and del(22q11) syndrome: a non-random association.

Authors:  A Verloes; C Curry; M Jamar; C Herens; P O'Lague; J Marks; P Sarda; P Blanchet
Journal:  J Med Genet       Date:  1998-11       Impact factor: 6.318

Review 5.  Excess of deletions of maternal origin in the DiGeorge/velo-cardio-facial syndromes. A study of 22 new patients and review of the literature.

Authors:  S Demczuk; A Lévy; M Aubry; M F Croquette; N Philip; M Prieur; U Sauer; P Bouvagnet; G A Rouleau; G Thomas
Journal:  Hum Genet       Date:  1995-07       Impact factor: 4.132

6.  Familial non-syndromic conotruncal defects are not associated with a 22q11 microdeletion.

Authors:  S Debrus; G Berger; A de Meeus; U Sauer; S Guillaumont; M Voisin; A Bozio; S Demczuk; A Aurias; P Bouvagnet
Journal:  Hum Genet       Date:  1996-02       Impact factor: 4.132

7.  Positional mapping of loci in the DiGeorge critical region at chromosome 22q11 using a new marker (D22S183).

Authors:  M P Mulder; M Wilke; A Langeveld; L G Wilming; A Hagemeijer; E van Drunen; E C Zwarthoff; P H Riegman; W H Deelen; A M van den Ouweland
Journal:  Hum Genet       Date:  1995-08       Impact factor: 4.132

Review 8.  Genomic approaches to identifying transcriptional regulators of osteoblast differentiation.

Authors:  Joseph P Stains; Roberto Civitelli
Journal:  Genome Biol       Date:  2003-07-01       Impact factor: 13.583

  8 in total

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