Os I, Aakesson I, Enger E
Acta Med Scand. 1985;217(4):429-34. doi: 10.1111/j.0954-6820.1985.tb02719.x.
A family comprising 46 members of 4 generations is described; 21 members suffered from incomplete diabetes insipidus (DI) of central origin. The pedigree showed a dominantly transmitted condition. The onset is gradual and starts in early infancy. The clinical symptoms are highly variable and decline in the sixth decade. Plasma vasopressin (AVP) during water deprivation was significantly lower in the DI group than in the controls (4.2 +/- 0.5 vs. 10.6 +/- 1.7 ng/l) (p less than 0.01), the difference being more pronounced in the high osmolality range (4.8 +/- 0.7 vs. 14.4 +/- 3.1 ng/l) (p less than 0.01). Urine osmolality was lower (241 +/- 36 vs. 928 +/- 46 mOsm/kg H2O) (p less than 0.01) despite higher serum osmolality during water deprivation, rendering the ratio between urine and serum osmolality less than unity compared with greater than 3:1 in the control group (p less than 0.001). In two affected females, addition of a non-osmotic stimulus caused no increase in plasma AVP. The findings are consistent with a partial defect in the production or release of AVP and not with a dysfunction of the intracranial osmoreceptors. The variable features of incomplete DI indicate that to define the condition by excessive urinary output alone is insufficient. The ratio between urine and serum osmolalities after an appropriate osmotic stimulus together with plasma AVP measurements may be necessary to confirm the diagnosis.
本文描述了一个包含4代46名成员的家族;其中21名成员患有中枢性不完全性尿崩症(DI)。该谱系显示为显性遗传疾病。发病较为隐匿,始于婴幼儿期。临床症状高度多变,在第六个十年会逐渐减轻。禁水期间,DI组血浆血管加压素(AVP)显著低于对照组(4.2±0.5对10.6±1.7 ng/l)(p<0.01),在高渗范围内差异更为明显(4.8±0.7对14.4±3.1 ng/l)(p<0.01)。尽管禁水期间血清渗透压较高,但尿渗透压较低(241±36对928±46 mOsm/kg H2O)(p<0.01),使得尿与血清渗透压之比小于1,而对照组大于3:1(p<0.001)。在两名受影响的女性中,非渗透性刺激并未导致血浆AVP升高。这些发现与AVP产生或释放的部分缺陷一致,而非颅内渗透压感受器功能障碍。不完全性DI的可变特征表明,仅通过尿量过多来定义该病是不够的。适当的渗透性刺激后尿与血清渗透压之比以及血浆AVP测量可能对于确诊是必要的。