Literature DB >> 10198163

Characterization and expression pattern of the frizzled gene Fzd9, the mouse homolog of FZD9 which is deleted in Williams-Beuren syndrome.

Y K Wang1, R Spörle, T Paperna, K Schughart, U Francke.   

Abstract

The frizzled gene family is conserved from insects to mammals and codes for putative Wnt receptors that share a cysteine-rich extracellular domain and seven transmembrane domains. We previously identified a novel frizzled gene, FZD3, now renamed FZD9, in the Williams-Beuren syndrome (WBS) deletion region at chromosomal band 7q11.23 and showed that its product can interact with the Drosophila wingless protein. Here, we report the characterization of the mouse homolog Fzd9. The Fzd9 gene produces a 2.4-kb transcript encoding a 592-amino-acid protein with 95% identity to the human FZD9. Fzd9 was mapped to the conserved syntenic region on distal mouse chromosome 5. By RNA in situ hybridization studies of whole-mount embryos and sections we delineated the temporal and spatial expression patterns in the neural tube, trunk skeletal muscle precursors (myotomes), limb skeletal anlagen, craniofacial regions, and nephric ducts. In adult mouse tissue, the Fzd9 transcript is abundantly present in heart, brain, testis, and skeletal muscle. In testis, Fzd9 is expressed in all spermatogenic cell types. Immunohistochemical studies of cells transfected with a Fzd9 expression construct confirm that Fzd9 is a membrane protein. These results suggest potential Wnt ligands of Fzd9, a role of Fzd9 in skeletal muscle specification, and contributions of FZD9 to the WBS phenotype. Copyright 1999 Academic Press.

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Year:  1999        PMID: 10198163     DOI: 10.1006/geno.1999.5773

Source DB:  PubMed          Journal:  Genomics        ISSN: 0888-7543            Impact factor:   5.736


  21 in total

1.  Generation and comparative analysis of approximately 3.3 Mb of mouse genomic sequence orthologous to the region of human chromosome 7q11.23 implicated in Williams syndrome.

Authors:  Udaya DeSilva; Laura Elnitski; Jacquelyn R Idol; Johannah L Doyle; Weiniu Gan; James W Thomas; Scott Schwartz; Nicole L Dietrich; Stephen M Beckstrom-Sternberg; Jennifer C McDowell; Robert W Blakesley; Gerard G Bouffard; Pamela J Thomas; Jeffrey W Touchman; Webb Miller; Eric D Green
Journal:  Genome Res       Date:  2002-01       Impact factor: 9.043

2.  A physical map, including a BAC/PAC clone contig, of the Williams-Beuren syndrome--deletion region at 7q11.23.

Authors:  R Peoples; Y Franke; Y K Wang; L Pérez-Jurado; T Paperna; M Cisco; U Francke
Journal:  Am J Hum Genet       Date:  2000-01       Impact factor: 11.025

3.  Wnt-1 regulation of connexin43 in cardiac myocytes.

Authors:  Z Ai; A Fischer; D C Spray; A M Brown; G I Fishman
Journal:  J Clin Invest       Date:  2000-01       Impact factor: 14.808

Review 4.  Frizzled homolog proteins, microRNAs and Wnt signaling in cancer.

Authors:  Koji Ueno; Hiroshi Hirata; Yuji Hinoda; Rajvir Dahiya
Journal:  Int J Cancer       Date:  2012-08-30       Impact factor: 7.396

5.  Essential roles of mesenchyme-derived beta-catenin in mouse Müllerian duct morphogenesis.

Authors:  Erica Deutscher; Humphrey Hung-Chang Yao
Journal:  Dev Biol       Date:  2007-05-03       Impact factor: 3.582

Review 6.  WNT/Frizzled signalling: receptor-ligand selectivity with focus on FZD-G protein signalling and its physiological relevance: IUPHAR Review 3.

Authors:  J P Dijksterhuis; J Petersen; G Schulte
Journal:  Br J Pharmacol       Date:  2014-03       Impact factor: 8.739

7.  Aberrant Wnt/β-Catenin Signaling Pathway in Testis of Azoospermic Men​.

Authors:  Marefat Ghaffari Novin; Reza Mirfakhraie; Hamid Nazarian
Journal:  Adv Pharm Bull       Date:  2015-09-19

Review 8.  A tale of two tracts: history, current advances, and future directions of research on sexual differentiation of reproductive tracts†.

Authors:  Fei Zhao; Humphrey Hung-Chang Yao
Journal:  Biol Reprod       Date:  2019-09-01       Impact factor: 4.285

9.  Induced chromosome deletions cause hypersociability and other features of Williams-Beuren syndrome in mice.

Authors:  Hong Hua Li; Madhuri Roy; Unsal Kuscuoglu; Corinne M Spencer; Birgit Halm; Katharine C Harrison; Joseph H Bayle; Alessandra Splendore; Feng Ding; Leslie A Meltzer; Elena Wright; Richard Paylor; Karl Deisseroth; Uta Francke
Journal:  EMBO Mol Med       Date:  2009-04       Impact factor: 12.137

10.  Physiopathological aspects of the Wnt/β-catenin signaling pathway in the male reproductive system.

Authors:  Ana Paola G Lombardi; Carine Royer; Raisa Pisolato; Fernanda N Cavalcanti; Thaís F G Lucas; Maria Fatima M Lazari; Catarina S Porto
Journal:  Spermatogenesis       Date:  2013-01-01
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