Hommes F A, Roesel R A, Metoki K, Hartlage P L, Dyken P R
Neuropediatrics. 1986 Feb;17(1):48-52. doi: 10.1055/s-2008-1052499.
A patient with the hyperornithinemia, hyperammonemia, homocitrullinuria syndrome is described. This patient represents the 12th documented case of this rare, presumably autosomal recessive condition. Increased levels of ammonia, ornithine and homocitrulline were demonstrated in blood and cerebrospinal fluid. The blood ammonia concentration could be lowered by supplementation of the diet with low doses of arginine. High doses of arginine precipitated seizures, although plasma levels of arginine and ornithine were not altered. The uptake of ornithine by the particulate fraction of the patient's fibroblasts was lower than that of controls, but still measurable. It is suggested that HHH patients have a partial impairment of the uptake of ornithine by mitochondria.
本文描述了一名患有高鸟氨酸血症、高氨血症、同型瓜氨酸尿综合征的患者。该患者是这种罕见的、可能为常染色体隐性疾病的第12例有记录的病例。血液和脑脊液中氨、鸟氨酸和同型瓜氨酸水平升高。通过在饮食中补充低剂量精氨酸可降低血氨浓度。尽管精氨酸和鸟氨酸的血浆水平未改变,但高剂量精氨酸会引发癫痫发作。患者成纤维细胞微粒体部分对鸟氨酸的摄取低于对照组,但仍可测量。提示HHH患者线粒体对鸟氨酸的摄取存在部分损害。