Perry T B, Holbrook K A, Hoff M S, Hamilton E F, Senikas V, Fisher C
Prenat Diagn. 1987 Mar;7(3):145-55. doi: 10.1002/pd.1970070302.
We report the first positive prenatal diagnosis of congenital non-bullous ichthyosiform erythroderma or lamellar ichthyosis. Fetal skin samples were obtained by fetoscopy at 21 weeks' gestation and examined by light and electron microscopy. Light microscopy revealed a thickened interfollicular epidermis with multiple layers of flattened cells and excessive keratinization of the epidermal lining of the follicular infundibulum. Electron microscopy of the thickened epidermis revealed granular cells that contained larger-than-normal keratohyalin granules and multiple layers of parakeratotic cornified cells. Although there was regional variation in the degree of interfollicular keratinization, follicles from all regions showed greater and more complete keratinization, indicating that they express the abnormality early enough in development to permit prenatal diagnosis at about 20 weeks' gestation.
我们报告了先天性非大疱性鱼鳞病样红皮病或板层状鱼鳞病的首例产前阳性诊断。在妊娠21周时通过胎儿镜检查获取胎儿皮肤样本,并进行光镜和电镜检查。光镜显示毛囊间表皮增厚,有多层扁平细胞,毛囊漏斗部表皮内衬过度角化。增厚表皮的电镜检查显示颗粒细胞含有大于正常的透明角质颗粒和多层不全角化的角质形成细胞。尽管毛囊间角化程度存在区域差异,但所有区域的毛囊均显示出更严重且更完全的角化,这表明它们在发育早期就表现出异常,从而能够在妊娠约20周时进行产前诊断。