Literature DB >> 15373767

Laminin-5 mutational analysis in an Italian cohort of patients with junctional epidermolysis bullosa.

Patrizia Posteraro1, Naomi De Luca, Guerrino Meneguzzi, May El Hachem, Corrado Angelo, Tommaso Gobello, Gianluca Tadini, Giovanna Zambruno, Daniele Castiglia.   

Abstract

Junctional epidermolysis bullosa (JEB) is a rare genodermatosis characterized by dermal-epidermal separation that is caused by mutations in the genes encoding hemidesmosomal components and laminin-5, the major epithelial adhesion ligand. Here, we report on the mutational analysis of LAMA3, LAMB3, and LAMC2 genes encoding laminin-5 chains in 19 Italian patients, 11 affected with the severe Herlitz (H JEB) and eight with the mild non-Herlitz variant of JEB (non-H JEB). Eighteen mutations, seven of which were novel, were identified and their consequences analyzed at the mRNA and protein level. Premature termination codon mutations in both alleles of LAMB3 or LAMC2 genes were found in nine of the 11 H JEB patients, with a prevalence of mutations in LAMC2. In one case, a homozygous frameshift mutation in LAMB3 was associated to illegitimate splicing leading to non-H JEB. One H JEB patient showed a large intragenic duplication within LAMC2, a genetic defect so far uncovered in laminin-5 genes. Splicing or missense mutations, were prevalent in non-H JEB patients. Collectively, five mutations appeared to be frequent in laminin-5 JEB patients: R635X, 29insC, E210K, W143X in LAMB3 and R95X in LAMC2. These recurrent mutations account for approximately 44% of laminin-5 JEB alleles in Italian patients.

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Year:  2004        PMID: 15373767     DOI: 10.1111/j.0022-202X.2004.23302.x

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


  7 in total

1.  Epidermolysis bullosa. I. Molecular genetics of the junctional and hemidesmosomal variants.

Authors:  R Varki; S Sadowski; E Pfendner; J Uitto
Journal:  J Med Genet       Date:  2006-02-10       Impact factor: 6.318

2.  CRISPR/Cas9-Mediated In Situ Correction of LAMB3 Gene in Keratinocytes Derived from a Junctional Epidermolysis Bullosa Patient.

Authors:  Daniela Benati; Francesca Miselli; Fabienne Cocchiarella; Clarissa Patrizi; Marta Carretero; Samantha Baldassarri; Virginia Ammendola; Cristina Has; Stefano Colloca; Marcela Del Rio; Fernando Larcher; Alessandra Recchia
Journal:  Mol Ther       Date:  2018-08-04       Impact factor: 11.454

Review 3.  Leading edge: emerging drug, cell, and gene therapies for junctional epidermolysis bullosa.

Authors:  Allison R Keith; Kirk Twaroski; Christen L Ebens; Jakub Tolar
Journal:  Expert Opin Biol Ther       Date:  2020-03-20       Impact factor: 4.388

4.  Clinical practice guidelines for laboratory diagnosis of epidermolysis bullosa.

Authors:  C Has; L Liu; M C Bolling; A V Charlesworth; M El Hachem; M J Escámez; I Fuentes; S Büchel; R Hiremagalore; G Pohla-Gubo; P C van den Akker; K Wertheim-Tysarowska; G Zambruno
Journal:  Br J Dermatol       Date:  2019-08-09       Impact factor: 9.302

5.  Recognizable neonatal clinical features of aplasia cutis congenita.

Authors:  Ingrid Anne Mandy Schierz; Mario Giuffrè; Antonello Del Vecchio; Vincenzo Antona; Giovanni Corsello; Ettore Piro
Journal:  Ital J Pediatr       Date:  2020-02-18       Impact factor: 2.638

6.  Proteasome-mediated degradation of keratins 7, 8, 17 and 18 by mutant KLHL24 in a foetal keratinocyte model: Novel insight in congenital skin defects and fragility of epidermolysis bullosa simplex with cardiomyopathy.

Authors:  Elena Logli; Elisa Marzuolo; Marco D'Agostino; Libenzio Adrian Conti; Anna Maria Lena; Andrea Diociaiuti; Elena Dellambra; Cristina Has; Valentina Cianfanelli; Giovanna Zambruno; May El Hachem; Alessandra Magenta; Eleonora Candi; Angelo Giuseppe Condorelli
Journal:  Hum Mol Genet       Date:  2022-04-22       Impact factor: 5.121

7.  A Novel Phenotype of Junctional Epidermolysis Bullosa with Transient Skin Fragility and Predominant Ocular Involvement Responsive to Human Amniotic Membrane Eyedrops.

Authors:  Daniele Castiglia; Paola Fortugno; Angelo Giuseppe Condorelli; Sabina Barresi; Naomi De Luca; Simone Pizzi; Iria Neri; Claudio Graziano; Diletta Trojan; Diego Ponzin; Sabrina Rossi; Giovanna Zambruno; Marco Tartaglia
Journal:  Genes (Basel)       Date:  2021-05-11       Impact factor: 4.096

  7 in total

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