Yang J M, Lee S, Kang H J, Lee J H, Yeo U C, Son I Y, Park K B, Steinert P M, Lee E S
Department of Dermatology, College of Medicine, Sungkyunkwan University, Samsung Medical Centre, Seoul, Korea.
Acta Derm Venereol. 1998 Nov;78(6):412-6. doi: 10.1080/000155598442674.
Palmoplantar keratodermas (PPK) constitute a heterogeneous group of diseases marked by the thickening of palms and soles of affected individuals. They are divided into autosomal dominant and autosomal recessive groups by the mode of transmission. The autosomal dominantly transmitted group is further divided into epidermolytic (EPPK, Voerner) and non-epidermolytic (NEPPK, Unna-Thost) types according to the histopathologic findings. Recent development of molecular approaches has confirmed that EPPK and NEPPK are caused by the mutations in keratin 9 and 1 genes, respectively. We have studied three families of EPPK to find the mutation in the keratin 9 gene. DNA sequence analyses revealed single base changes in sequences encoding the highly conserved 1A rod domain segment of the keratin 9 gene in two of the three families. These mutations caused Arg (CGG) to Glu (CAG; R162Q) and Arg (CGG) to Try (TGG; R162W) substitutions. The same arginine position has been mutated in the keratin 10 gene in epidermolytic hyperkeratosis, the keratin 14 gene in epidermolysis bullosa simplex, and the keratin 9 gene in hereditary EPPK in Western patients. In this study we show that unrelated Korean patients have similar mutations.
掌跖角化病(PPK)是一组异质性疾病,其特征是受累个体的手掌和脚底增厚。根据遗传方式,它们分为常染色体显性遗传和常染色体隐性遗传两组。常染色体显性遗传组根据组织病理学结果进一步分为表皮松解性(EPPK,沃纳型)和非表皮松解性(NEPPK,乌纳 - 托斯特型)。分子生物学方法的最新进展证实,EPPK和NEPPK分别由角蛋白9和1基因的突变引起。我们研究了三个EPPK家族,以寻找角蛋白9基因中的突变。DNA序列分析显示,在三个家族中的两个家族中,编码角蛋白9基因高度保守的1A杆状结构域片段的序列发生了单碱基变化。这些突变导致精氨酸(CGG)被谷氨酸(CAG;R162Q)取代以及精氨酸(CGG)被色氨酸(TGG;R162W)取代。在西方患者的表皮松解性角化过度中的角蛋白10基因、单纯性大疱性表皮松解症中的角蛋白14基因以及遗传性EPPK中的角蛋白9基因中,相同的精氨酸位置发生了突变。在本研究中,我们表明不相关的韩国患者有相似的突变。