Literature DB >> 24965254

ZRS 406A>G mutation in patients with tibial hypoplasia, polydactyly and triphalangeal first fingers.

Phatchara Norbnop1, Chalurmpon Srichomthong2, Kanya Suphapeetiporn2, Vorasuk Shotelersuk2.   

Abstract

Werner mesomelic syndrome (WMS), an autosomal dominant disorder characterized by hypoplastic tibiae, triphalangeal thumbs and polydactyly, is caused by a specific point mutation at the position 404 in zone of polarizing activity regulatory sequence (ZRS). Here we identified two additional families with WMS. All three patients in three generations of Family 1 were found to harbor the same heterozygous 406A>G mutation in ZRS. The fourth patient from Family 2 was a sporadic case with the known 404 point mutation. The novel 406A>G mutation expands mutational spectrum in ZRS causing WMS, provides evidence for a functionally important nucleotide position 406 of ZRS in humans and has implications for genetic counseling.

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Year:  2014        PMID: 24965254     DOI: 10.1038/jhg.2014.50

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  19 in total

Review 1.  Tibial hemimelia-polydactyly-five-fingered hand syndrome associated with a 404 G>A mutation in a distant sonic hedgehog cis-regulator (ZRS): a case report.

Authors:  Tae-Joon Cho; Goo Hyun Baek; Hey-Ran Lee; Hyuk Joo Moon; Won Joon Yoo; In Ho Choi
Journal:  J Pediatr Orthop B       Date:  2013-05       Impact factor: 1.041

2.  HnRNP U mediates the long-range regulation of Shh expression during limb development.

Authors:  Jing Zhao; Jun Ding; Yingqian Li; Kaiqun Ren; Jiahao Sha; Minsheng Zhu; Xiang Gao
Journal:  Hum Mol Genet       Date:  2009-05-28       Impact factor: 6.150

Review 3.  Growing models of vertebrate limb development.

Authors:  Matthew Towers; Cheryll Tickle
Journal:  Development       Date:  2009-01       Impact factor: 6.868

4.  Prenatal whole-genome sequencing--is the quest to know a fetus's future ethical?

Authors:  Ilana R Yurkiewicz; Bruce R Korf; Lisa Soleymani Lehmann
Journal:  N Engl J Med       Date:  2014-01-16       Impact factor: 91.245

5.  A series of ENU-induced single-base substitutions in a long-range cis-element altering Sonic hedgehog expression in the developing mouse limb bud.

Authors:  Hiroshi Masuya; Hideki Sezutsu; Yoshiyuki Sakuraba; Tomoko Sagai; Masaki Hosoya; Hideki Kaneda; Ikuo Miura; Kimio Kobayashi; Kenta Sumiyama; Aya Shimizu; Junko Nagano; Haruka Yokoyama; Satoko Kaneko; Noriko Sakurai; Yuka Okagaki; Tetsuo Noda; Shigeharu Wakana; Yoichi Gondo; Toshihiko Shiroishi
Journal:  Genomics       Date:  2006-10-13       Impact factor: 5.736

6.  A long-range Shh enhancer regulates expression in the developing limb and fin and is associated with preaxial polydactyly.

Authors:  Laura A Lettice; Simon J H Heaney; Lorna A Purdie; Li Li; Philippe de Beer; Ben A Oostra; Debbie Goode; Greg Elgar; Robert E Hill; Esther de Graaff
Journal:  Hum Mol Genet       Date:  2003-07-15       Impact factor: 6.150

7.  Triphalangeal thumb and five-fingered hand.

Authors:  K M Chan; D W Lamb
Journal:  Hand       Date:  1983-10

8.  Phylogenetic conservation of a limb-specific, cis-acting regulator of Sonic hedgehog ( Shh).

Authors:  Tomoko Sagai; Hiroshi Masuya; Masaru Tamura; Kunihiko Shimizu; Yukari Yada; Shigeharu Wakana; Yoichi Gondo; Tetsuo Noda; Toshihiko Shiroishi
Journal:  Mamm Genome       Date:  2004-01       Impact factor: 2.957

9.  A specific mutation in the distant sonic hedgehog (SHH) cis-regulator (ZRS) causes Werner mesomelic syndrome (WMS) while complete ZRS duplications underlie Haas type polysyndactyly and preaxial polydactyly (PPD) with or without triphalangeal thumb.

Authors:  Dagmar Wieczorek; Barbara Pawlik; Yun Li; Nurten A Akarsu; Almuth Caliebe; Klaus J W May; Bernd Schweiger; Fernando R Vargas; Sevim Balci; Gabriele Gillessen-Kaesbach; Bernd Wollnik
Journal:  Hum Mutat       Date:  2010-01       Impact factor: 4.878

10.  Opposing functions of the ETS factor family define Shh spatial expression in limb buds and underlie polydactyly.

Authors:  Laura A Lettice; Iain Williamson; John H Wiltshire; Silvia Peluso; Paul S Devenney; Alison E Hill; Abdelkader Essafi; James Hagman; Richard Mort; Graeme Grimes; Carlo L DeAngelis; Robert E Hill
Journal:  Dev Cell       Date:  2012-02-14       Impact factor: 12.270

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  7 in total

1.  Comprehensive In Vivo Interrogation Reveals Phenotypic Impact of Human Enhancer Variants.

Authors:  Evgeny Z Kvon; Yiwen Zhu; Guy Kelman; Catherine S Novak; Ingrid Plajzer-Frick; Momoe Kato; Tyler H Garvin; Quan Pham; Anne N Harrington; Riana D Hunter; Janeth Godoy; Eman M Meky; Jennifer A Akiyama; Veena Afzal; Stella Tran; Fabienne Escande; Brigitte Gilbert-Dussardier; Nolwenn Jean-Marçais; Sanjarbek Hudaiberdiev; Ivan Ovcharenko; Matthew B Dobbs; Christina A Gurnett; Sylvie Manouvrier-Hanu; Florence Petit; Axel Visel; Diane E Dickel; Len A Pennacchio
Journal:  Cell       Date:  2020-03-12       Impact factor: 41.582

Review 2.  Postaxial limb hypoplasia (PALH): the classification, clinical features, and related developmental biology.

Authors:  Zeng Zhang; Dan Yi; Rong Xie; John L Hamilton; Qing-Lin Kang; Di Chen
Journal:  Ann N Y Acad Sci       Date:  2017-10-09       Impact factor: 5.691

3.  The Conserved Sonic Hedgehog Limb Enhancer Consists of Discrete Functional Elements that Regulate Precise Spatial Expression.

Authors:  Laura A Lettice; Paul Devenney; Carlo De Angelis; Robert E Hill
Journal:  Cell Rep       Date:  2017-08-08       Impact factor: 9.423

Review 4.  Zone of Polarizing Activity Regulatory Sequence Mutations/Duplications with Preaxial Polydactyly and Longitudinal Preaxial Ray Deficiency in the Phenotype: A Review of Human Cases, Animal Models, and Insights Regarding the Pathogenesis.

Authors:  Mohammad M Al-Qattan
Journal:  Biomed Res Int       Date:  2018-02-13       Impact factor: 3.411

Review 5.  A multidisciplinary review of triphalangeal thumb.

Authors:  Jacob W P Potuijt; Robert-Jan H Galjaard; Peter J van der Spek; Christianne A van Nieuwenhoven; Nadav Ahituv; Kerby C Oberg; Steven E R Hovius
Journal:  J Hand Surg Eur Vol       Date:  2018-10-14

6.  Association of SNP rs80659072 in the ZRS with polydactyly in Beijing You chickens.

Authors:  Qin Chu; Zhixun Yan; Jian Zhang; Tahir Usman; Yao Zhang; Hui Liu; Haihong Wang; Ailian Geng; Huagui Liu
Journal:  PLoS One       Date:  2017-10-09       Impact factor: 3.240

7.  Variable expression of subclinical phenotypes instead of reduced penetrance in families with mild triphalangeal thumb phenotypes.

Authors:  Jacob W P Potuijt; Jeannette Hoogeboom; Esther de Graaff; Christianne A van Nieuwenhoven; Robert Jan H Galjaard
Journal:  J Med Genet       Date:  2020-03-16       Impact factor: 6.318

  7 in total

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