van Dijk M A, Chang P C, Peters D J, Breuning M H
Department of Nephrology, University Hospital Leiden, Netherlands.
J Am Soc Nephrol. 1995 Dec;6(6):1670-3. doi: 10.1681/ASN.V661670.
Autosomal dominant polycystic kidney disease is genetically heterogenous, with at least two chromosomal loci accounting for the disease. When the mutation is located on chromosome 16 (PKD1), extra-renal manifestations such as the rupture of intracranial aneurysms are well known. In the case of localization on chromosome 4 (PKD2), in which the renal disease runs a milder course, not much is known about the incidence of extrarenal manifestations. A PKD2 family is reported in which two members had subarachnoidal bleeding due to intracranial aneurysms; there was strong clinical evidence of subarachnoidal bleeding in a third family member. This indicates that the familial clustering of intracranial aneurysms may also occur in PKD2 families. Because of the considerable mortality and morbidity of intracranial aneurysms, screening with magnetic resonance angiography in PKD2 patients with a positive family history of intracranial aneurysms is recommended.
常染色体显性多囊肾病在遗传上具有异质性,至少有两个染色体位点与该病有关。当突变位于16号染色体(PKD1)时,肾外表现如颅内动脉瘤破裂是众所周知的。而当定位在4号染色体(PKD2)时,肾病进程相对较轻,关于肾外表现的发生率所知甚少。本文报道了一个PKD2家系,其中两名成员因颅内动脉瘤导致蛛网膜下腔出血;第三名家族成员有蛛网膜下腔出血的有力临床证据。这表明颅内动脉瘤的家族聚集性也可能发生在PKD2家系中。由于颅内动脉瘤具有相当高的死亡率和发病率,建议对有颅内动脉瘤家族史阳性的PKD2患者进行磁共振血管造影筛查。