Bettinelli A, Bianchetti M G, Borella P, Volpini E, Metta M G, Basilico E, Selicorni A, Bargellini A, Grassi M R
Istituto Clinica Pediatrica II, Univesritá di Milano, Italy.
Kidney Int. 1995 Feb;47(2):547-51. doi: 10.1038/ki.1995.68.
To better clarify the genetic inheritance of primary tubular hypomagnesemia-hypokalemia with hypocalciuria, or Gitelman's syndrome (GS), we studied eight families (10 patients aged 11 to 22 years; 16 parents; 9 siblings) in which at least one offspring had GS (plasma magnesium < 0.65 mmol/liter; plasma potassium < 3.6 mmol/liter; high magnesium and potassium fractional excretions; molar urinary calcium/creatinine < 0.10). Two families each had two offspring of different sex with GS, who all had tetanic episodes and/or marked weakness during childhood or adolescence, whereas in three other families two mothers and three offspring presented GS and one father and two other offspring had hypomagnesemia and hypocalciuria but normal plasma potassium. The mean plasma magnesium and potassium levels of the patients of the first two families were significantly lower (P < 0.05) than those of the other three families. Intralymphocytic but not intraerythrocytic magnesium and potassium were significantly lower (P < 0.05) in patients compared to controls. We hypothesize that there are two different types of genetic transmission of GS, one autosomal recessive and one autosomal dominant with high phenotype variability. It seems that this genetic heterogeneity is associated with a different clinical expression with frequent tetanic episodes and lower plasma potassium and magnesium levels in the autosomal recessive form.
为了更清楚地阐明原发性肾小管低镁血症 - 低血钾伴低钙尿症(即吉特林综合征,GS)的遗传方式,我们研究了8个家系(10例患者,年龄在11至22岁之间;16名父母;9名兄弟姐妹),其中至少有一个后代患有GS(血浆镁<0.65 mmol/升;血浆钾<3.6 mmol/升;镁和钾的分数排泄率高;尿钙/肌酐摩尔比<0.10)。两个家系各有两名不同性别的后代患有GS,他们在儿童期或青春期均有手足搐搦发作和/或明显虚弱,而在另外三个家系中,两名母亲和三名后代患有GS,一名父亲和另外两名后代有低镁血症和低钙尿症,但血浆钾正常。前两个家系患者的平均血浆镁和钾水平显著低于(P<0.05)其他三个家系。与对照组相比,患者淋巴细胞内而非红细胞内的镁和钾显著降低(P<0.05)。我们推测GS存在两种不同的遗传传递类型,一种是常染色体隐性遗传,另一种是常染色体显性遗传且表型变异性高。这种遗传异质性似乎与不同的临床表型有关,常染色体隐性遗传形式中手足搐搦发作频繁,血浆钾和镁水平较低。