Kurajoh Masafumi, Koyama Hidenori, Hatayama Miki, Okazaki Hirokazu, Shoji Takuhito, Moriwaki Yuji, Yamamoto Tetsuya, Nakayama Tomitaka, Namba Mitsuyoshi
Division of Diabetes, Endocrinology and Metabolism, Department of Internal Medicine, Hyogo College of Medicine, Japan.
Intern Med. 2015;54(12):1523-6. doi: 10.2169/internalmedicine.54.3290. Epub 2015 Jun 15.
A 15-year-old boy was referred to our department due to gout. The laboratory findings showed hyperuricemia with a decreased erythrocyte hypoxanthine phosphoribosyl transferase (HPRT) activity. The HPRT cDNA sequence was revealed to be 206A>T, which has not been previously reported. In addition, direct sequencing of genomic DNA showed the patient to possess four variants reported to be associated with hyperuricemia. This is the first case report of partial HPRT deficiency due to a novel HPRT mutation accompanied by variants associated with hyperuricemia. Combination treatment consisting of benzbromarone and febuxostat had a significant effect in reducing the urate level in our patient.
一名15岁男孩因痛风转诊至我科。实验室检查结果显示高尿酸血症,红细胞次黄嘌呤磷酸核糖转移酶(HPRT)活性降低。HPRT cDNA序列显示为206A>T,此前未见报道。此外,基因组DNA直接测序显示该患者存在4种据报道与高尿酸血症相关的变异。这是首例因新型HPRT突变伴高尿酸血症相关变异导致部分HPRT缺乏的病例报告。苯溴马隆和非布司他联合治疗对降低该患者尿酸水平有显著效果。