Literature DB >> 31197392

[Syndromes with skin fragility].

A Reimer1, C Has2.   

Abstract

Syndromic disorders with skin fragility belong to different groups of genodermatoses: epidermolysis bullosa (EB), Ehlers-Danlos syndrome and porphyria. The genetic defects mainly concern structural proteins which assure the mechanical stability of the skin and other tissues. Depending on the expression pattern of the affected protein in the skin, cutaneous fragility may manifest as superficial erosions, blisters, wounds, wound healing defects or scars. Extracutaneous manifestations are manifold and involve the heart, skeletal muscles, intestine, kidneys, blood vessels or the skeleton. Syndromic types of EB include in addition to skin blistering: (i) cardiomyopathy in case of desmoplakin, plakoglobin, or kelch-like protein mutations; (ii) muscular dystrophy in case of plektin mutations; (iii) pyloric atresia in case of integrin α6β4 or plectin mutations; (iv) nephrotic syndrome in case of CD151 or integrin α3 mutations. Lysyl hydroxylase 3 mutations affect posttranslational modifications of collagens and lead to a dystrophic epidermolysis bullosa-like multisystemic disorder. Ehlers-Danlos syndromes are due to defects of dermal collagens or their processing and affect the skin, joints and blood vessels. Finally porphyrias are complex metabolic disorders with photosensitivity and sometimes skin fragility, liver or neurologic problems. Their pathogenesis relies on the accumulation of precursors in the tissues. Although these syndromes are rare in clinical practice, knowledge of the syndromic constellation contributes to early diagnosis and detection of complications.

Entities:  

Keywords:  Ehlers-Danlos syndrome; Epidermolysis bullosa; Genodermatoses; Porphyrias; Pyloric atresia

Mesh:

Year:  2019        PMID: 31197392     DOI: 10.1007/s00105-019-4433-5

Source DB:  PubMed          Journal:  Hautarzt        ISSN: 0017-8470            Impact factor:   0.751


  38 in total

1.  PLEC1 mutations underlie adult-onset dilated cardiomyopathy in epidermolysis bullosa simplex with muscular dystrophy.

Authors:  Maria C Bolling; Hendri H Pas; Marianne de Visser; Eleonora Aronica; Ellen G Pfendner; Maarten P van den Berg; Gilles F H Diercks; Albert J H Suurmeijer; Marcel F Jonkman
Journal:  J Invest Dermatol       Date:  2009-12-17       Impact factor: 8.551

Review 2.  Skin and heart: une liaison dangereuse.

Authors:  Maria C Bolling; Marcel F Jonkman
Journal:  Exp Dermatol       Date:  2009-06-23       Impact factor: 3.960

3.  Plectin gene mutations can cause epidermolysis bullosa with pyloric atresia.

Authors:  Ellen Pfendner; Jouni Uitto
Journal:  J Invest Dermatol       Date:  2005-01       Impact factor: 8.551

4.  Erythropoietic protoporphyria: a family study and report of a novel mutation in the FECH gene.

Authors:  Paulo Morais; Alberto Mota; Teresa Baudrier; Filipe Trigo; João Paulo Oliveira; Rita Cerqueira; Aida Palmeiro; Purificação Tavares; Filomena Azevedo
Journal:  Eur J Dermatol       Date:  2011 Jul-Aug       Impact factor: 3.328

Review 5.  Porphyrias.

Authors:  Hervé Puy; Laurent Gouya; Jean-Charles Deybach
Journal:  Lancet       Date:  2010-03-13       Impact factor: 79.321

Review 6.  Genetic aspects of porphyria cutanea tarda.

Authors:  Richard W Lambrecht; Manish Thapar; Herbert L Bonkovsky
Journal:  Semin Liver Dis       Date:  2007-02       Impact factor: 6.115

7.  Plectin expression patterns determine two distinct subtypes of epidermolysis bullosa simplex.

Authors:  Ken Natsuga; Wataru Nishie; Masashi Akiyama; Hideki Nakamura; Satoru Shinkuma; James R McMillan; Akari Nagasaki; Cristina Has; Takeshi Ouchi; Akira Ishiko; Yoshiaki Hirako; Katsushi Owaribe; Daisuke Sawamura; Leena Bruckner-Tuderman; Hiroshi Shimizu
Journal:  Hum Mutat       Date:  2010-03       Impact factor: 4.878

8.  Reduction of lysyl hydroxylase 3 causes deleterious changes in the deposition and organization of extracellular matrix.

Authors:  Maija Risteli; Heli Ruotsalainen; Antti M Salo; Raija Sormunen; Laura Sipilä; Naomi L Baker; Shireen R Lamandé; Leena Vimpari-Kauppinen; Raili Myllylä
Journal:  J Biol Chem       Date:  2009-08-20       Impact factor: 5.157

9.  CD151, the first member of the tetraspanin (TM4) superfamily detected on erythrocytes, is essential for the correct assembly of human basement membranes in kidney and skin.

Authors:  Vanja Karamatic Crew; Nicholas Burton; Alexander Kagan; Carole A Green; Cyril Levene; Frances Flinter; R Leo Brady; Geoff Daniels; David J Anstee
Journal:  Blood       Date:  2004-07-20       Impact factor: 22.113

10.  A connective tissue disorder caused by mutations of the lysyl hydroxylase 3 gene.

Authors:  Antti M Salo; Helen Cox; Peter Farndon; Celia Moss; Helen Grindulis; Maija Risteli; Simon P Robins; Raili Myllylä
Journal:  Am J Hum Genet       Date:  2008-10-02       Impact factor: 11.025

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