Reis A, Hennies H C, Langbein L, Digweed M, Mischke D, Drechsler M, Schröck E, Royer-Pokora B, Franke W W, Sperling K
Institute of Human Genetics, Free University, Berlin, Germany.
Nat Genet. 1994 Feb;6(2):174-9. doi: 10.1038/ng0294-174.
We have isolated the gene for human type I keratin 9 (KRT9) and localised it to chromosome 17q21. Patients with epidermolytic palmoplantar keratoderma (EPPK), an autosomal dominant skin disease, were investigated. Three KRT9 mutations, N160K, R162Q, and R162W, were identified. All the mutations are in the highly conserved coil 1A of the rod domain, thought to be important for heterodimerisation. R162W was detected in five unrelated families and affects the corresponding residue in the keratin 14 and keratin 10 genes that is also altered in cases of epidermolysis bullosa simplex and generalised epidermolytic hyperkeratosis, respectively. These findings provide further evidence that mutations in keratin genes may cause epidermolysis and hyperkeratosis and that hyperkeratosis of palms and soles may be caused by different mutations in the KRT9 gene.
我们已经分离出人类I型角蛋白9(KRT9)基因,并将其定位到17号染色体的q21区域。对患有常染色体显性遗传性皮肤病——表皮松解性掌跖角化病(EPPK)的患者进行了研究。鉴定出三个KRT9突变,分别为N160K、R162Q和R162W。所有突变均位于杆状结构域高度保守的卷曲1A区域,该区域被认为对异源二聚化很重要。在五个无亲缘关系的家族中检测到R162W突变,该突变影响角蛋白14和角蛋白10基因中的相应残基,而在单纯性大疱性表皮松解症和全身性表皮松解性角化过度症病例中,这两个基因中的相应残基也分别发生了改变。这些发现进一步证明,角蛋白基因突变可能导致表皮松解和角化过度,并且掌跖角化过度可能由KRT9基因中的不同突变引起。