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致死性(赫利茨)交界性大疱性表皮松解症中,层粘连蛋白5(LAMA3)α3链基因的纯合无义突变。

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

作者信息

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

机构信息

Department of Dermatology, Jefferson Medical College, Philadelphia, PA, USA.

出版信息

Hum Mol Genet. 1995 May;4(5):959-62. doi: 10.1093/hmg/4.5.959.

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.

摘要

遗传性机械性大疱性疾病——交界性大疱性表皮松解症(JEB)的特征是皮肤和黏膜出现广泛水疱和糜烂。JEB的诊断标志包括皮肤基底膜带半桥粒的超微结构异常,以及用抗锚定细丝蛋白层粘连蛋白5的抗体染色阴性。因此,编码层粘连蛋白5的α3、β3和γ2链的三个基因,即LAMA3、LAMB3和LAMC2,是JEB的候选基因。我们之前已在几个患有JEB的家族中证实了LAMB3和LAMC2基因存在突变。我们从一名患病儿童开始进行突变分析,通过对LAMA3各个外显子进行PCR扩增,随后进行异源双链分析。对异源双链进行核苷酸测序,在α3链的I/II结构域内发现了一个纯合无义突变。这些发现首次证明LAMA3基因内的无义突变也参与了JEB的发病机制,并表明层粘连蛋白5的所有三个基因的突变均可导致JEB表型。

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