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层粘连蛋白-5(LAMB3)β3链编码基因中的E210K突变可预测泛发性萎缩性良性大疱性表皮松解症的表型。

E210K mutation in the gene encoding the beta3 chain of laminin-5 (LAMB3) is predictive of a phenotype of generalized atrophic benign epidermolysis bullosa.

作者信息

Mellerio J E, Eady R A, Atherton D J, Lake B D, McGrath J A

机构信息

Department of Cell and Molecular Pathology, St John's Institute of Dermatology (UMDS), St Thomas' Hospital, London SE1 7EH, U.K.

出版信息

Br J Dermatol. 1998 Aug;139(2):325-31. doi: 10.1046/j.1365-2133.1998.02377.x.

DOI:10.1046/j.1365-2133.1998.02377.x
PMID:9767254
Abstract

Pathogenetic mutations in the genes encoding the hemidesmosome-anchoring filament complex proteins, laminin-5 and the 180 kDa bullous pemphigoid antigen, have been identified in patients with the inherited mechanobullous disease, junctional epidermolysis bullosa (EB). Furthermore, there is some evidence to suggest that precise definition of the nature of mutations in these genes may correlate to specific phenotypes of disease. We report three junctional EB patients who carry an identical missense mutation, E210K, on one allele of the gene encoding the beta3 subunit chain of laminin-5 (LAMB3) in addition to different nonsense mutations on the second allele. Two of the patients are adults and display a specific phenotype of non-lethal junctional EB known as generalized atrophic benign EB, which is associated with trauma-induced blisters, nail dystrophy and alopecia. As the third patient is a young child with fewer features of this subtype to date, identification of E210K in combination with a nonsense LAMB3 mutation may be predictive of the subsequent development of a generalized atrophic benign EB phenotype both in this child and in other junctional EB patients with the E210K mutation. Identification of this particular mutation has important implications for clinical management and counselling.

摘要

在遗传性机械性大疱病——交界性大疱性表皮松解症(EB)患者中,已鉴定出编码半桥粒锚定细丝复合物蛋白、层粘连蛋白-5和180 kDa大疱性类天疱疮抗原的基因中的致病突变。此外,有证据表明,这些基因中突变性质的精确界定可能与疾病的特定表型相关。我们报告了三名交界性EB患者,他们在编码层粘连蛋白-5(LAMB3)β3亚基链的基因的一个等位基因上携带相同的错义突变E210K,同时在第二个等位基因上携带不同的无义突变。其中两名患者为成年人,表现出一种名为泛发性萎缩性良性EB的非致命性交界性EB的特定表型,该表型与创伤性水疱、甲营养不良和脱发有关。由于第三名患者是一名幼儿,迄今为止该亚型的特征较少,因此在该儿童以及其他携带E210K突变的交界性EB患者中,鉴定出E210K与LAMB3无义突变相结合可能预示着泛发性萎缩性良性EB表型的后续发展。鉴定出这一特定突变对临床管理和咨询具有重要意义。

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E210K mutation in the gene encoding the beta3 chain of laminin-5 (LAMB3) is predictive of a phenotype of generalized atrophic benign epidermolysis bullosa.层粘连蛋白-5(LAMB3)β3链编码基因中的E210K突变可预测泛发性萎缩性良性大疱性表皮松解症的表型。
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