Literature DB >> 18239616

Unique and recurrent mutations in the filaggrin gene in Singaporean Chinese patients with ichthyosis vulgaris.

Huijia Chen1, Jean C C Ho, Aileen Sandilands, Yuin Chew Chan, Yoke Chin Giam, Alan T Evans, E Birgitte Lane, W H Irwin McLean.   

Abstract

Filaggrin is an abundant protein of the outer epidermis that is essential for terminal differentiation of keratinocytes and formation of an effective barrier against water loss and pathogen/allergen/irritant invasion. Recent investigations in Europe and Japan have revealed null mutations in the filaggrin gene (FLG) as the underlying cause of ichthyosis vulgaris (IV), a common skin disorder characterised by dry skin, palmar hyperlinearity and keratosis pilaris. Following the development of a strategy for the comprehensive analysis of FLG, we have identified five unique mutations and one recurrent mutation in Singaporean Chinese IV patients. Mutation 441delA is located in the profilaggrin S100 domain, whereas two additional frameshift mutations, 1249insG and 7945delA, occur in the first partial filaggrin repeat ("repeat 0") and in filaggrin repeat 7, respectively. Both nonsense mutations Q2147X and E2422X are found in filaggrin repeat 6, whereas R4307X was found on one of the longer size variant alleles of FLG, within duplicated repeat 10.2. Mutation E2422X, previously found in a single Dutch patient, was found in one Singaporean IV patient and at a low frequency in Asian population controls. Our study confirms the presence of population-specific as well as recurrent FLG mutations in Singapore.

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Year:  2008        PMID: 18239616     DOI: 10.1038/sj.jid.2008.2

Source DB:  PubMed          Journal:  J Invest Dermatol        ISSN: 0022-202X            Impact factor:   8.551


  12 in total

1.  Genetic Polymorphism of FLG in Korean Ichthyosis Vulgaris Patients.

Authors:  Eun Joo Kim; Mi Sook Jeong; Kapsok Li; Mi Kyung Park; Mi-Kyung Lee; Yoosik Yoon; Dae-Yeon Cho; Seong Jun Seo
Journal:  Ann Dermatol       Date:  2011-05-27       Impact factor: 1.444

Review 2.  [Epidermal barrier disorders in dermatoses].

Authors:  E Proksch; D Dähnhardt; R Fölster-Holst
Journal:  Hautarzt       Date:  2016-11       Impact factor: 0.751

3.  Filaggrin in the frontline: role in skin barrier function and disease.

Authors:  Aileen Sandilands; Calum Sutherland; Alan D Irvine; W H Irwin McLean
Journal:  J Cell Sci       Date:  2009-05-01       Impact factor: 5.285

Review 4.  One remarkable molecule: filaggrin.

Authors:  Sara J Brown; W H Irwin McLean
Journal:  J Invest Dermatol       Date:  2011-12-08       Impact factor: 8.551

5.  Moisturisers from birth in at-risk infants of atopic dermatitis - a pragmatic randomised controlled trial.

Authors:  Pamela Si Min Ng; Lynette Wei Yi Wee; Valerie Pui Yoong Ho; Weixuan Colin Tan; Priya Bishnoi; Uma Alagappan; Sharon Mun Yee Wong; Emily Yiping Gan; Bin Huey Quek; Liang Shen; Bing Su; John Ea Common; Mark Jean Aan Koh
Journal:  Australas J Dermatol       Date:  2021-08-23       Impact factor: 2.481

Review 6.  Natural history and risk factors of atopic dermatitis in children.

Authors:  Bok Yang Pyun
Journal:  Allergy Asthma Immunol Res       Date:  2014-11-25       Impact factor: 5.764

7.  Study protocol to investigate the environmental and genetic aetiology of atopic dermatitis: the Indonesian Prospective Study of Atopic Dermatitis in Infants (ISADI).

Authors:  Conny Tanjung; Peter Rzehak; Muchtaruddin Mansyur; Zakiudin Munasir; Herawati Sudoyo; Suzanna Immanuel; Roedi Irawan; Eva Reischl; Hans Demmelmair; Berthold Koletzko; Sri Rezeki Hadinegoro; Damayanti Rusli Sjarif
Journal:  BMJ Open       Date:  2017-03-03       Impact factor: 2.692

8.  Filaggrin haploinsufficiency is highly penetrant and is associated with increased severity of eczema: further delineation of the skin phenotype in a prospective epidemiological study of 792 school children.

Authors:  S J Brown; C L Relton; H Liao; Y Zhao; A Sandilands; W H I McLean; H J Cordell; N J Reynolds
Journal:  Br J Dermatol       Date:  2009-06-11       Impact factor: 9.302

9.  Mutations in the Filaggrin are Predisposing Factor in Korean Children With Atopic Dermatitis.

Authors:  Ho-Sung Yu; Mi-Jin Kang; Young-Ho Jung; Hyung-Young Kim; Ju-Hee Seo; Young-Joon Kim; Seung-Hwa Lee; Ha-Jung Kim; Ji-Won Kwon; Byoung-Ju Kim; Jinho Yu; Soo-Jong Hong
Journal:  Allergy Asthma Immunol Res       Date:  2013-03-20       Impact factor: 5.764

10.  Unique variants in the FLG gene and FERMT1 gene in a Chinese patient with ichthyosis and Kindler syndrome.

Authors:  Bo-Wen Zheng; Xian-Zhong Zhu; Yu Lan; Jian-Chi Ma; Xi-Qing Li
Journal:  JAAD Case Rep       Date:  2019-11-22
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