Hovnanian A, de Prost Y
Service de dermatologie, hôpital Necker-Enfants-Malades, Paris, France.
Arch Pediatr. 1994 Nov;1(11):1028-33.
Inherited epidermolysis bullosa (EB) includes three main types depending on the precise ultrastructural level at which the split responsible for the blistering occurs; 1) simplex EB (SEB) located at the level of the basal cells; 2) junctional EB (JEB) located in the lamina lucida; 3) dystrophic EB (DEB) located in the dermis below the lamina densa at the level of the anchoring fibrils. The authors review the major recent progresses which have led to the identification of the genes and of several molecular defects in these three types, such as: mutations of the genes of keratins 5 and 14 in SEB, molecular defects in the gamma 2 chain gene of nicein/kalinin coding for the anchoring fibrils in JEB, abnormalities of the collagen VII gene coding for the anchoring fibrils in DEB. These data allow to consider a classification of EB based on molecular defects. They also have important consequences for genetic counselling and prenatal diagnosis for the families presenting an affected child.
遗传性大疱性表皮松解症(EB)根据水疱形成所涉及的分裂发生的精确超微结构水平主要分为三种类型:1)单纯型EB(SEB),分裂发生在基底细胞水平;2)交界型EB(JEB),位于透明层;3)营养不良型EB(DEB),位于致密层下方真皮中的锚原纤维水平。作者回顾了近期的主要进展,这些进展促成了这三种类型中相关基因及多种分子缺陷的鉴定,例如:SEB中角蛋白5和14基因的突变、JEB中编码锚原纤维的巢蛋白/卡利宁γ2链基因的分子缺陷、DEB中编码锚原纤维的胶原蛋白VII基因的异常。这些数据使得基于分子缺陷对EB进行分类成为可能。它们对于有患病儿童的家庭的遗传咨询和产前诊断也具有重要意义。