Literature DB >> 7633458

A homozygous nonsense mutation in the alpha 3 chain gene of laminin 5 (LAMA3) in lethal (Herlitz) junctional epidermolysis bullosa.

S Kivirikko1, J A McGrath, C Baudoin, D Aberdam, S Ciatti, M G Dunnill, J R McMillan, R A Eady, J P Ortonne, G Meneguzzi.   

Abstract

The inherited mechanobullous disorder, junctional epidermolysis bullosa (JEB), is characterized by extensive blistering and erosions of the skin and mucous membranes. The diagnostic hallmarks of JEB include ultrastructural abnormalities in the hemidesmosomes of the cutaneous basement membrane zone, as well as an absence of staining with antibodies against the anchoring filament protein, laminin 5. Therefore, the three genes encoding alpha 3, beta 3 and gamma 2 chains of laminin 5, known as LAMA3, LAMB3 and LAMC2, are candidate genes for JEB. We have previously demonstrated mutations in the LAMB3 and LAMC2 genes in several families with JEB. We initiated mutation analysis from an affected child by PCR amplification of individual LAMA3 exons, followed by heteroduplex analysis. Nucleotide sequencing of heteroduplexes identified a homozygous nonsense mutation within domain I/II of the alpha 3 chain. These findings provide the first evidence that nonsense mutations within the LAMA3 gene are also involved in the pathogenesis of JEB, and indicate that mutations of all three genes of laminin 5 can result in the JEB phenotype.

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Year:  1995        PMID: 7633458     DOI: 10.1093/hmg/4.5.959

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  30 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

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Review 4.  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

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Journal:  Adv Genomics Genet       Date:  2015-06-12

6.  Recessive Mutations in ACPT, Encoding Testicular Acid Phosphatase, Cause Hypoplastic Amelogenesis Imperfecta.

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Review 7.  The genetics of human skin disease.

Authors:  Gina M DeStefano; Angela M Christiano
Journal:  Cold Spring Harb Perspect Med       Date:  2014-10-01       Impact factor: 6.915

8.  Localization of laminin alpha3B chain in vascular and epithelial basement membranes of normal human tissues and its down-regulation in skin cancers.

Authors:  Yoshinobu Kariya; Taizo Mori; Chie Yasuda; Naoko Watanabe; Yoshie Kaneko; Yukiko Nakashima; Takashi Ogawa; Kaoru Miyazaki
Journal:  J Mol Histol       Date:  2008-08-01       Impact factor: 2.611

Review 9.  The molecular basis for inherited bullous diseases.

Authors:  B P Korge; T Krieg
Journal:  J Mol Med (Berl)       Date:  1996-02       Impact factor: 4.599

Review 10.  Molecular genetics of ameloblast cell lineage.

Authors:  Marianna Bei
Journal:  J Exp Zool B Mol Dev Evol       Date:  2009-07-15       Impact factor: 2.656

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