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遗传性大疱性表皮松解症(Herlitz交界型)中,层粘连蛋白5(LAMB3)的β3链基因存在纯合性无义突变。

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

作者信息

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

机构信息

Department of Dermatology, Jefferson Medical College, Thomas Jefferson University, Philadelphia, Pennsylvania 19107.

出版信息

Genomics. 1994 Nov 15;24(2):357-60. doi: 10.1006/geno.1994.1627.

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.

摘要

赫利茨交界型大疱性表皮松解症(H-JEB)是一种严重的常染色体隐性疾病,其特征是在真皮-表皮基底膜内形成水疱。基于识别层粘连蛋白5表位(以前称为巢蛋白/卡利宁)的免疫荧光分析,这种透明层蛋白的基因已被提议作为H-JEB的候选基因。在本研究中,我们通过对一个患有H-JEB的家族中的先证者角质形成细胞mRNA进行Northern杂交和RT-PCR分析,检测了编码层粘连蛋白5β3多肽链的基因(LAMB3)。Northern分析显示层粘连蛋白β3链mRNA水平显著降低。通过RT-PCR扩增mRNA,随后进行直接核苷酸测序,发现两个等位基因均发生了纯合的C到T转换,导致提前终止密码子(CGA→TGA)。该突变在基因组DNA水平得到验证,且父母均显示为相同突变的杂合携带者。这是首次对H-JEB患者中层粘连蛋白5的层粘连蛋白β3链基因(LAMB3)突变的描述。

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