Literature DB >> 26288439

Ectodermal Dysplasia-Skin Fragility Syndrome: A Rare Case Report.

Subhash Kashyap1, Vinay Shanker1, Neelam Sharma2.   

Abstract

Ectodermal dysplasia/skin fragility syndrome (ED-SFS) is a newly described autosomal recessive disorder characterized by skin fragility and blistering, palmoplantar keratoderma, abnormal hair growth, nail dystrophy, and occasionally defective sweating. It results from mutations in the PKP1 gene encoding plakophilin 1 (PKP1), which is an important component of stratifying epithelial desmosomes and a nuclear component of many cell types. Only 12 cases of this rare genodermatosis have been reported so far. We present an unusual case of ED-SFS in a 12-year boy who was normal at birth but subsequently developed skin fragility, hair and nail deformities, abnormal dentition, palmoplantar keratoderma, and abnormal sweating but no systemic abnormality.

Entities:  

Keywords:  Chelitis; McGrath syndrome; ectodermal dysplasia/skin fragility syndrome; granulosis rubra nasi; palmoplantar keratoderma

Year:  2015        PMID: 26288439      PMCID: PMC4533569          DOI: 10.4103/0019-5154.160525

Source DB:  PubMed          Journal:  Indian J Dermatol        ISSN: 0019-5154            Impact factor:   1.494


  10 in total

1.  Carboxyl terminus of Plakophilin-1 recruits it to plasma membrane, whereas amino terminus recruits desmoplakin and promotes desmosome assembly.

Authors:  Tammy Sobolik-Delmaire; Dawn Katafiasz; James K Wahl
Journal:  J Biol Chem       Date:  2006-04-21       Impact factor: 5.157

2.  The classification of inherited epidermolysis bullosa (EB): Report of the Third International Consensus Meeting on Diagnosis and Classification of EB.

Authors:  Jo-David Fine; Robin A J Eady; Eugene A Bauer; Johann W Bauer; Leena Bruckner-Tuderman; Adrian Heagerty; Helmut Hintner; Alain Hovnanian; Marcel F Jonkman; Irene Leigh; John A McGrath; Jemima E Mellerio; Dedee F Murrell; Hiroshi Shimizu; Jouni Uitto; Anders Vahlquist; David Woodley; Giovanna Zambruno
Journal:  J Am Acad Dermatol       Date:  2008-04-18       Impact factor: 11.527

3.  Ectodermal dysplasia-skin fragility syndrome.

Authors:  Vijay S Adhe; Atul M Dongre; Uday S Khopkar
Journal:  Indian J Dermatol Venereol Leprol       Date:  2011 Jul-Aug       Impact factor: 2.545

4.  Ectodermal dysplasia-skin fragility syndrome resulting from a new homozygous mutation, 888delC, in the desmosomal protein plakophilin 1.

Authors:  Sibel Ersoy-Evans; Gül Erkin; Hiva Fassihi; Ien Chan; Amy S Paller; Selçuk Sürücü; John A McGrath
Journal:  J Am Acad Dermatol       Date:  2006-07       Impact factor: 11.527

5.  Genotype-phenotype correlation in skin fragility-ectodermal dysplasia syndrome resulting from mutations in plakophilin 1.

Authors:  T Hamada; A P South; Y Mitsuhashi; T Kinebuchi; O Bleck; G H S Ashton; Y Hozumi; T Suzuki; T Hashimoto; R A J Eady; J A McGrath
Journal:  Exp Dermatol       Date:  2002-04       Impact factor: 3.960

6.  The distribution of the desmosomal protein, plakophilin 1, in human skin and skin tumors.

Authors:  I Moll; H Kurzen; L Langbein; W W Franke
Journal:  J Invest Dermatol       Date:  1997-02       Impact factor: 8.551

Review 7.  Ectodermal dysplasia-skin fragility syndrome.

Authors:  John A McGrath; Jemima E Mellerio
Journal:  Dermatol Clin       Date:  2010-01       Impact factor: 3.478

8.  Novel truncating mutations in PKP1 and DSP cause similar skin phenotypes in two Brazilian families.

Authors:  A Tanaka; J E Lai-Cheong; M E M Café; B Gontijo; P R Salomão; L Pereira; J A McGrath
Journal:  Br J Dermatol       Date:  2008-10-21       Impact factor: 9.302

Review 9.  Plakophilin 1: an important stabilizer of desmosomes.

Authors:  A P South
Journal:  Clin Exp Dermatol       Date:  2004-03       Impact factor: 3.470

10.  Oral rehabilitation of a patient with ectodermal dysplasia with prosthodontics treatment.

Authors:  Srinivas Nallanchakrava
Journal:  Indian J Dermatol       Date:  2013-05       Impact factor: 1.494

  10 in total

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