Lemire J, Kaplan B S
Am J Nephrol. 1984;4(2):81-5. doi: 10.1159/000166782.
Three phenotypic forms of cystinosis (nephropathic, juvenile-onset and benign adult forms) are well recognized. That not all infants and children have the classical nephropathic form with Fanconi's syndrome is illustrated by 2 of the 3 patients in this report. Case 1 had the typical nephropathic form with generalized proximal tubular dysfunction. In contrast, case 2 had transient features of Bartter's syndrome which preceded the manifestations of Fanconi's syndrome. Case 3 was more complex because he first presented with a syndrome of nephrogenic diabetes insipidus. He then developed features of Fanconi's syndrome as well as metabolic acidosis, hyponatremia and hyperkalemia. The failure of deoxycorticosteroid administration to elevate the serum concentration suggested, in retrospect, that he may also have had a form of acquired pseudohypoaldosteronism.
胱氨酸病的三种表型形式(肾病型、青少年型和良性成人型)已得到充分认识。本报告中的3例患者中有2例表明,并非所有婴幼儿都患有伴有范科尼综合征的典型肾病型。病例1具有伴有广泛性近端肾小管功能障碍的典型肾病型。相比之下,病例2在范科尼综合征表现之前有巴特综合征的短暂特征。病例3更为复杂,因为他最初表现为肾性尿崩症综合征。随后他出现了范科尼综合征的特征以及代谢性酸中毒、低钠血症和高钾血症。回顾来看,脱氧皮质类固醇给药未能提高血清浓度,提示他可能还患有某种获得性假性醛固酮增多症。