Berson E L, Shih V E, Sullivan P L
Ophthalmology. 1981 Apr;88(4):311-5. doi: 10.1016/s0161-6420(81)35029-5.
Five patients, ages 12 to 30, with gyrate atrophy have shown substantial (60% or greater) decreases in plasma ornithine concentrations within four to eight weeks when placed on a therapeutic trial of low-protein (10-15 g/day), low-arginine diets supplemented with essential amino acids (EAA) and pyridoxine hydrochloride. Four of five patients have continued on modified protein restriction (20-35 g/day) and one on pyridoxine (300 mg/day) alone with maintenance of plasma ornithine in the range of 30 to 60% below pretherapeutic trial levels. After one year, four of five patients have shown no significant improvement in visual acuity, fields, final dark-adapted thresholds, electroretinograms, or fundus appearance. One patient with the poorest control of plasma ornithine has developed a decrease in ERG amplitudes and a new area of chorioretinal atrophy. These patients continue in this trial to determine whether or not any reductions in hyperornithinemia will modify the course of the ocular disease.
5名年龄在12至30岁之间的回旋状萎缩患者,在接受低蛋白(10 - 15克/天)、低精氨酸饮食并补充必需氨基酸(EAA)和盐酸吡哆醇的治疗试验后,4至8周内血浆鸟氨酸浓度大幅下降(60%或更高)。5名患者中有4名继续接受改良的蛋白质限制饮食(20 - 35克/天),1名仅服用盐酸吡哆醇(300毫克/天),血浆鸟氨酸维持在治疗试验前水平以下30%至60%的范围内。一年后,5名患者中有4名在视力、视野、最终暗适应阈值、视网膜电图或眼底外观方面没有显著改善。血浆鸟氨酸控制最差的一名患者视网膜电图振幅降低,出现了新的脉络膜视网膜萎缩区域。这些患者继续参与该试验,以确定高鸟氨酸血症的任何降低是否会改变眼部疾病的病程。