Literature DB >> 7698759

A homozygous nonsense mutation in the beta 3 chain gene of laminin 5 (LAMB3) in Herlitz junctional epidermolysis bullosa.

L Pulkkinen1, A M Christiano, D Gerecke, D W Wagman, R E Burgeson, M R Pittelkow, J Uitto.   

Abstract

Herlitz junctional epidermolysis bullosa (H-JEB) is a severe autosomal recessive disorder characterized by blister formation within the dermal-epidermal basement membrane. Based on immunofluorescence analysis recognizing laminin 5 epitopes (previously known as nicein/kalinin), the genes for this lamina lucida protein have been proposed as candidate genes in H-JEB. In this study, we examined the gene encoding the beta 3 polypeptide chain of laminin 5 (LAMB3) by Northern hybridization and RT-PCR analysis of keratinocyte mRNA from a proband in a family with H-JEB. Northern analysis revealed markedly reduced levels of the laminin beta 3 chain mRNA. Amplification of mRNA by RT-PCR, followed by direct nucleotide sequencing, revealed a homozygous C-to-T transition resulting in a premature termination codon (CGA --> TGA) on both alleles. This mutation was verified at the genomic DNA level, and both parents were shown to be heterozygous carriers of the same mutation. This is the first description of a mutation in the laminin beta 3 chain gene (LAMB3) of laminin 5 in an H-JEB patient.

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Year:  1994        PMID: 7698759     DOI: 10.1006/geno.1994.1627

Source DB:  PubMed          Journal:  Genomics        ISSN: 0888-7543            Impact factor:   5.736


  44 in total

1.  IAP insertion in the murine LamB3 gene results in junctional epidermolysis bullosa.

Authors:  J E Kuster; M H Guarnieri; J G Ault; L Flaherty; P J Swiatek
Journal:  Mamm Genome       Date:  1997-09       Impact factor: 2.957

2.  Clinical assessment incorporating a personal genome.

Authors:  Euan A Ashley; Atul J Butte; Matthew T Wheeler; Rong Chen; Teri E Klein; Frederick E Dewey; Joel T Dudley; Kelly E Ormond; Aleksandra Pavlovic; Alexander A Morgan; Dmitry Pushkarev; Norma F Neff; Louanne Hudgins; Li Gong; Laura M Hodges; Dorit S Berlin; Caroline F Thorn; Katrin Sangkuhl; Joan M Hebert; Mark Woon; Hersh Sagreiya; Ryan Whaley; Joshua W Knowles; Michael F Chou; Joseph V Thakuria; Abraham M Rosenbaum; Alexander Wait Zaranek; George M Church; Henry T Greely; Stephen R Quake; Russ B Altman
Journal:  Lancet       Date:  2010-05-01       Impact factor: 79.321

3.  Maternal uniparental disomy of chromosome 1 with reduction to homozygosity of the LAMB3 locus in a patient with Herlitz junctional epidermolysis bullosa.

Authors:  L Pulkkinen; F Bullrich; P Czarnecki; L Weiss; J Uitto
Journal:  Am J Hum Genet       Date:  1997-09       Impact factor: 11.025

4.  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

5.  Deletion of the laminin alpha4 chain leads to impaired microvessel maturation.

Authors:  Jill Thyboll; Jarkko Kortesmaa; Renhai Cao; Raija Soininen; Ling Wang; Antti Iivanainen; Lydia Sorokin; Mårten Risling; Yihai Cao; Karl Tryggvason
Journal:  Mol Cell Biol       Date:  2002-02       Impact factor: 4.272

Review 6.  The role of laminins in basement membrane function.

Authors:  M Aumailley; N Smyth
Journal:  J Anat       Date:  1998-07       Impact factor: 2.610

7.  Dynamic interactions of epidermal collagen XVII with the extracellular matrix: laminin 332 as a major binding partner.

Authors:  Wataru Nishie; Dimitra Kiritsi; Alexander Nyström; Silke C Hofmann; Leena Bruckner-Tuderman
Journal:  Am J Pathol       Date:  2011-06-14       Impact factor: 4.307

Review 8.  Lessons from skin blistering: molecular mechanisms and unusual patterns of inheritance?

Authors:  A S Paller
Journal:  Am J Pathol       Date:  1996-06       Impact factor: 4.307

9.  Prenatal diagnosis for recessive dystrophic epidermolysis bullosa in 10 families by mutation and haplotype analysis in the type VII collagen gene (COL7A1).

Authors:  A M Christiano; S LaForgia; A S Paller; J McGuire; H Shimizu; J Uitto
Journal:  Mol Med       Date:  1996-01       Impact factor: 6.354

10.  Revertant mosaicism in junctional epidermolysis bullosa due to multiple correcting second-site mutations in LAMB3.

Authors:  Anna M G Pasmooij; Hendri H Pas; Maria C Bolling; Marcel F Jonkman
Journal:  J Clin Invest       Date:  2007-05       Impact factor: 14.808

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