Eilers E, Stieler K, Thies C, Blume-Peytavi U, Obladen M, Hüseman D
Klinik für Neonatologie, Campus Virchow Klinik, Charité Universitätsmedizin Berlin, Augustenburger Platz 1, Berlin.
Klin Padiatr. 2010 Mar;222(2):86-69. doi: 10.1055/s-0029-1220943. Epub 2010 Feb 17.
Harlequin ichthyosis is the most severe congenital keratinizing disorder. It is caused by mutations in the ABCA12 gene leading to defective lipid transport. The infants are born with ectropion, eclabium and fissured plate-like skin. Today these infants can survive with neonatal intensive care and retinoid therapy and need long-term interdisciplinary treatment in order to improve quality of life. However, the outcome in our case is impaired by severe psychomotor developmental delay, which has not yet been associated with Harlequin Ichthyosis.
丑角样鱼鳞病是最严重的先天性角化障碍。它由ABCA12基因突变引起,导致脂质转运缺陷。婴儿出生时伴有睑外翻、唇外翻和板层状皮肤皲裂。如今,这些婴儿通过新生儿重症监护和维甲酸治疗能够存活,并且需要长期的多学科治疗以提高生活质量。然而,我们这个病例的结果受到严重精神运动发育迟缓的影响,这种情况尚未与丑角样鱼鳞病相关联。