Meisel A D, Diamond H S
Am J Med Sci. 1977 Jan-Feb;273(1):109-15. doi: 10.1097/00000441-197701000-00013.
A patient with asymptomatic adult Fanconi syndrome with glycosuria, amino-aciduria, hypophosphatemia, and renal tubular acidosis was found to have hypouricemia (serum uric acid, 1.5-1.8 mg/100 ml) secondary to increased renal clearance of urate (urate clearance/glomerular filtration rate, 32 per cent). Increased urate clearance in this patient with multiple reabsorptive defects probably represents diminished urate reabsorption. Consistent with this, the uricosuric response to probenecid was diminished. Reabsorption of filtered urate appeared to be intact. Inhibition of urate secretion with pyrazinamide completely suppressed the uricosuria in this patient, suggesting that increased urate clearance was due to either enhanced secretion or diminished reabsorption of secreted urate. There was no direct evidence for increased urate secretion. The response of urate excretion to pharmacologic inhibitors of tubular transport of urate differs in various clinical states associated with hyperuricosuria. The responses may reflect different mechanisms of hyperuricosuria.
一名患有无症状成人范科尼综合征的患者,伴有糖尿、氨基酸尿、低磷血症和肾小管酸中毒,被发现继发于尿酸肾清除率增加(尿酸清除率/肾小球滤过率,32%)而出现低尿酸血症(血清尿酸,1.5 - 1.8 mg/100 ml)。该患有多种重吸收缺陷的患者尿酸清除率增加可能代表尿酸重吸收减少。与此一致的是,对丙磺舒的促尿酸排泄反应减弱。滤过尿酸的重吸收似乎完好无损。用吡嗪酰胺抑制尿酸分泌完全抑制了该患者的尿酸尿,表明尿酸清除率增加是由于分泌增强或分泌尿酸的重吸收减少。没有直接证据表明尿酸分泌增加。在与高尿酸尿相关的各种临床状态下,尿酸排泄对尿酸肾小管转运的药理抑制剂的反应有所不同。这些反应可能反映了高尿酸尿的不同机制。