Rodes M, Ribes A, Pineda M, Alvarez L, Fabregas I, Fernandez Alvarez E, Coude F X, Grimber G
J Inherit Metab Dis. 1987;10(1):73-81. doi: 10.1007/BF01799492.
We have reported three cases of hyperornithinaemia associated with hyperammonaemia and homocitrullinuria (HHH). They deal with two brothers and a sister from a family where the parents and four other children are healthy on clinical and biochemical examination. The biochemical findings in our patients indicate the existence of a defect in the transport of ornithine into the mitochondria. Cultured skin fibroblasts from two of these patients incorporated six times less [14C]ornithine into protein as compared to control cells. The most characteristic sign of the clinical picture is the progressive spastic paraparesis found in one of the cases. Ornithine supplementation and restricted protein intake may be useful in the treatment of this syndrome since after three years of treatment the clinical response was favourable and the patients showed no adverse clinical effects.
我们报告了3例与高氨血症和高鸟氨酸血症相关的同型瓜氨酸尿症(HHH)。病例涉及一个家庭中的两兄弟和一姐妹,该家庭的父母及其他四个孩子经临床和生化检查均健康。我们患者的生化检查结果表明存在鸟氨酸向线粒体转运的缺陷。其中两名患者的培养皮肤成纤维细胞与对照细胞相比,将[14C]鸟氨酸掺入蛋白质的量减少了六倍。临床症状最典型的体征是其中一例出现的进行性痉挛性截瘫。补充鸟氨酸和限制蛋白质摄入可能对该综合征的治疗有用,因为经过三年治疗,临床反应良好,患者未出现不良临床影响。