Kovarik J, Vormittag W, Gebhart W, Ruthner U, Lubec G, Molzer B
Wien Klin Wochenschr. 1978 Dec 8;90(23):839-44.
Mucopolysaccharidosis V (Scheie's syndrome, MPS-IS) is a very rare, autosomal recessively inherited metabolic disease. The degradation of dermatan sulphate and heparan sulphate is disturbed due to alpha-L-iduronidase deficiency, leading to intracellular storage and excessive urinary secretion of these substances. The characteristic clinical features are contractures (claw-like flexion of the fingers), umbilical and inguinal herniae, corneal opacity, hepatomegaly, myocardiopathy and minor skeletal malformations. A patient with Scheie's syndrome is now reported for the first time in Austria; the results of the clinical, biochemical, chromosomal, dermatoglyphic and electron optical investigations are described and discussed.
黏多糖贮积症Ⅴ型(施艾氏综合征,MPS-IS)是一种非常罕见的常染色体隐性遗传代谢疾病。由于α-L-艾杜糖醛酸酶缺乏,硫酸皮肤素和硫酸乙酰肝素的降解受到干扰,导致这些物质在细胞内蓄积并经尿液过量分泌。其特征性临床症状为挛缩(手指呈爪状屈曲)、脐疝和腹股沟疝、角膜混浊、肝肿大、心肌病以及轻微骨骼畸形。本文首次报道了奥地利一名施艾氏综合征患者;并描述和讨论了临床、生化、染色体、皮纹和电子光学检查结果。