Department of Pediatrics and Research Laboratory for Mitochondrial Disorders, Ajou University School of Medicine, Suwon, Korea.
J Korean Med Sci. 2010 Jan;25(1):172-5. doi: 10.3346/jkms.2010.25.1.172. Epub 2009 Dec 26.
Cystinuria is an inherited renal and intestinal disease characterized by defective amino acids reabsorption and cystine urolithiasis. It is unusually associated with neurologic symptoms. Mutations in two genes, SLC3A1 and SLC7A9, have been identified in cystinuric patients. This report presents a 13-yr-old boy with cystinuria who manifested difficulty in walking, ataxia, and mental retardation. Somatosensory evoked potential of posterior tibial nerve stimulation showed the central conduction dysfunction through the posterior column of spinal cord. He was diagnosed non-type I cystinuria by urinary amino acid analysis and oral cystine loading test. We screened him and his family for gene mutation by direct sequencing of SLC3A1 and SLC7A9 genes. In this patient, we identified new missence mutation G173R in SLC7A9 gene.
胱氨酸尿症是一种遗传性肾脏和肠道疾病,其特征为氨基酸吸收缺陷和胱氨酸尿结石。它异常伴有神经系统症状。在胱氨酸尿症患者中已鉴定出两个基因 SLC3A1 和 SLC7A9 的突变。本报告介绍了一名 13 岁男孩,他患有胱氨酸尿症,表现为行走困难、共济失调和智力迟钝。刺激后胫神经体感诱发电位显示通过脊髓后柱的中枢传导功能障碍。通过尿氨基酸分析和口服胱氨酸负荷试验,他被诊断为非 I 型胱氨酸尿症。我们通过直接测序 SLC3A1 和 SLC7A9 基因对他及其家人进行基因突变筛查。在该患者中,我们在 SLC7A9 基因中发现了新的错义突变 G173R。