Prabahar M Ram, Manorajan R, Fernando M E, Venkatraman R, Balaraman V, Jayakumar M
Department of Nephrology, Madras Medical College and Government General Hospital, Chennai.
J Assoc Physicians India. 2006 Jun;54:497-500.
Familial Hypomagnesemia, Hypercalciuria with Nephrocalcinosis is a rare autosomal recessive inherited disease associated with renal failure. Two girls born of consanguineous parentage aged 16 and 17 presented to us with renal failure, nephrocalcinosis and bone deformities. On evaluation they were found to have hypomagnesemia, hypercalciuria, increased fractional excretion of magnesium, hypocitraturia, renal failure and elevated PTH. Their parental screening was normal. There were no extra-renal features in them. One sibling had nephrolithiasis and the stone analysis revealed calcium phosphate stones. Both were treated with sodium bicarbonate, thiazides, calcitriol and calcium carbonate. They did not require dialysis during hospital stay. Both of them were treated conservatively. They are on regular outpatient follow up. The primary defect in this syndrome is impaired paracellular reabsorption of magnesium and calcium in the medullary thick ascending limb. Mutations in the PCLN-1gene which encodes for the tight junction protein paracellin -1 is identified as the underlying genetic defect. Ocular abnormalities and deafness are the commonly reported associations. End stage renal failure usually occurs in second to third decade. Renal transplantation is the definite treatment.
家族性低镁血症、高钙尿症伴肾钙质沉着症是一种与肾衰竭相关的罕见常染色体隐性遗传病。两名来自近亲父母的16岁和17岁女孩因肾衰竭、肾钙质沉着症和骨骼畸形前来就诊。经评估,她们存在低镁血症、高钙尿症、镁排泄分数增加、低枸橼酸尿症、肾衰竭和甲状旁腺激素升高。其父母筛查正常。她们没有肾外表现。其中一名同胞有肾结石,结石分析显示为磷酸钙结石。两人均接受了碳酸氢钠、噻嗪类利尿剂、骨化三醇和碳酸钙治疗。住院期间她们无需透析。两人均接受保守治疗。她们定期门诊随访。该综合征的主要缺陷是髓袢升支粗段镁和钙的细胞旁重吸收受损。编码紧密连接蛋白claudin -1的PCLN -1基因突变被确定为潜在的遗传缺陷。眼部异常和耳聋是常见的相关表现。终末期肾衰竭通常发生在第二至第三个十年。肾移植是明确的治疗方法。