Literature DB >> 29307792

Further delineation of the SOX18-related Hypotrichosis, Lymphedema, Telangiectasia syndrome (HTLS).

Irene Valenzuela1, Paula Fernández-Alvarez2, Alberto Plaja2, Gema Ariceta3, Anna Sabaté-Rotés4, Elena García-Arumí5, Teresa Vendrell2, Eduardo Tizzano6.   

Abstract

The transcription factor SOX18 has been shown to play a role in the development of hair, blood and lymphatic vessels. Mutations in SOX18 result in hereditary lymphedema, with the unique clinical association of hypotrichosis and telangiectasia (HLTS). Some patients present with additional disease features which may be explained by the location of SOX18 mutation. We report a patient with hypotrichosis-lymphedema-telangiectasia syndrome (HLTS) confirmed by detection of a novel mutation in the SOX18 gene. Few cases of HTLS have been reported in the literature. We reviewed all cases reported to date to delineate the clinical manifestations that allow us to prompt diagnosis of this syndrome for appropriate management and genetic counseling.
Copyright © 2018 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  C3 glomerulonephritis; Hypotrichosis; Lymphedema; SOX18; Telangiectasia

Mesh:

Substances:

Year:  2018        PMID: 29307792     DOI: 10.1016/j.ejmg.2018.01.001

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  6 in total

1.  Homodimerization regulates an endothelial specific signature of the SOX18 transcription factor.

Authors:  Mehdi Moustaqil; Frank Fontaine; Jeroen Overman; Alex McCann; Timothy L Bailey; Paulina Rudolffi Soto; Akshay Bhumkar; Nichole Giles; Dominic J B Hunter; Yann Gambin; Mathias Francois; Emma Sierecki
Journal:  Nucleic Acids Res       Date:  2018-11-30       Impact factor: 16.971

2.  A dominant-negative SOX18 mutant disrupts multiple regulatory layers essential to transcription factor activity.

Authors:  Alex J McCann; Jieqiong Lou; Mehdi Moustaqil; Matthew S Graus; Ailisa Blum; Frank Fontaine; Hui Liu; Winnie Luu; Paulina Rudolffi-Soto; Peter Koopman; Emma Sierecki; Yann Gambin; Frédéric A Meunier; Zhe Liu; Elizabeth Hinde; Mathias Francois
Journal:  Nucleic Acids Res       Date:  2021-11-08       Impact factor: 16.971

3.  The SOXE transcription factors-SOX8, SOX9 and SOX10-share a bi-partite transactivation mechanism.

Authors:  Abdul Haseeb; Véronique Lefebvre
Journal:  Nucleic Acids Res       Date:  2019-07-26       Impact factor: 16.971

Review 4.  Sorting Sox: Diverse Roles for Sox Transcription Factors During Neural Crest and Craniofacial Development.

Authors:  Elizabeth N Schock; Carole LaBonne
Journal:  Front Physiol       Date:  2020-12-08       Impact factor: 4.566

5.  R-propranolol is a small molecule inhibitor of the SOX18 transcription factor in a rare vascular syndrome and hemangioma.

Authors:  Jeroen Overman; Frank Fontaine; Jill Wylie-Sears; Mehdi Moustaqil; Lan Huang; Marie Meurer; Ivy Kim Chiang; Emmanuelle Lesieur; Jatin Patel; Johannes Zuegg; Eddy Pasquier; Emma Sierecki; Yann Gambin; Mohamed Hamdan; Kiarash Khosrotehrani; Gregor Andelfinger; Joyce Bischoff; Mathias Francois
Journal:  Elife       Date:  2019-07-30       Impact factor: 8.140

6.  Genome-Wide Selective Signatures Reveal Candidate Genes Associated with Hair Follicle Development and Wool Shedding in Sheep.

Authors:  Zhihui Lei; Weibo Sun; Tingting Guo; Jianye Li; Shaohua Zhu; Zengkui Lu; Guoyan Qiao; Mei Han; Hongchang Zhao; Bohui Yang; Liping Zhang; Jianbin Liu; Chao Yuan; Yaojing Yue
Journal:  Genes (Basel)       Date:  2021-11-29       Impact factor: 4.096

  6 in total

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