Castronuovo S, Simon J W, Kandel G L, Morier A, Wolf B, Witkop C J, Jenkins P L
Department of Ophthalmology, Albany Medical College, New York 12208.
Am J Ophthalmol. 1991 Apr 15;111(4):419-26. doi: 10.1016/s0002-9394(14)72374-8.
We studied the albinotic characteristics in 13 members of a white family (age range, 2 to 73 years), which were graded according to severity and were correlated with visual acuity. Clinical, electrophysiologic, and biochemical characteristics of this family do not fit any known category of human albinism. The degree of heterogeneity in expression of albinotic features was unexpected. The correlation between visual acuity and nystagmus was particularly strong. The brown-haired propositus had severe skin involvement, iris transillumination, fundus hypopigmentation, and foveal hypoplasia. He had no manifest nystagmus, however, and his visual acuity was nearly normal. These observations suggest that nystagmus imposes a visual deficit beyond that related to foveal hypoplasia alone.
我们研究了一个白人家庭中13名成员(年龄范围为2至73岁)的白化病特征,这些特征根据严重程度分级,并与视力相关。该家庭的临床、电生理和生化特征不符合任何已知的人类白化病类别。白化病特征表达的异质性程度出乎意料。视力与眼球震颤之间的相关性尤为显著。这位棕色头发的先证者有严重的皮肤受累、虹膜透照、眼底色素减退和黄斑发育不全。然而,他没有明显的眼球震颤,并且视力几乎正常。这些观察结果表明,眼球震颤造成的视力缺陷不仅仅与单纯的黄斑发育不全有关。