Vabres P, Larrégue M
INSERM U393, Hôpital Necker-Enfants Malades, Paris.
Ann Dermatol Venereol. 1995;122(4):154-60.
Chromosome X is one of the best genetically defined. Many disease loci are assigned to this chromosome, due to the peculiar mode of inheritance of X-linked disorders. Chromosome X undergoes X-inactivation in females. Recombination with chromosome Y occurs at pseudoautosomal regions. Some features of X-linked genodermatoses are a consequence of these phenomenons: variable expression, topography following Blaschko's lines. This can be seen in incontinentia pigmenti, focal dermal hypoplasia or hypohidrotic ectodermal dysplasia. Deletions at the pseudoautosomal region may cause contiguous gene syndromes. Hence ichthyosis with steroid-sulfatase deficiency may occur in association with various disorders. Transmitting females should be recognized by clinical examination or molecular studies, as this represents the main point in genetic counselling.
X染色体是遗传定义最清晰的染色体之一。由于X连锁疾病独特的遗传方式,许多疾病基因座都定位于这条染色体上。在女性中,X染色体会发生X失活。与Y染色体的重组发生在假常染色体区域。X连锁遗传性皮肤病的一些特征就是这些现象导致的:表达可变、沿布拉斯科线分布。这在色素失禁症、局灶性真皮发育不全或少汗性外胚层发育不良中都可以看到。假常染色体区域的缺失可能会导致相邻基因综合征。因此,类固醇硫酸酯酶缺乏症鱼鳞病可能与各种疾病相关。临床检查或分子研究应识别出携带致病基因的女性,因为这是遗传咨询的要点。