Elchediak Daniel S, Cahill Anne Marie, Furth Emma E, Kaplan Bernard S, Hartung Erum A
aEmory University, Atlanta, Georgia, USA.
bDepartment of Radiology, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
Case Rep Nephrol Dial. 2017 May 2;7(2):34-42. doi: 10.1159/000475492. eCollection 2017 May-Aug.
Unlike autosomal dominant polycystic kidney disease (ADPKD), autosomal recessive polycystic kidney disease (ARPKD) is not generally known to be associated with vascular abnormalities. Only 4 cases of ARPKD patients with intracranial aneurysms have been reported previously. We present 2 ARPKD patients with extracranial vascular abnormalities: a young man with infrarenal aortic and iliac artery aneurysms complicated by dissection and a teenage girl with multiple splenic and gastric artery aneurysms and arterial vascular malformations. These cases raise the question of whether vascular integrity and development may be impaired in ARPKD, perhaps through molecular mechanisms overlapping with ADPKD. This possibility is supported by studies in mice that show ARPKD gene expression in the walls of large blood vessels.
与常染色体显性多囊肾病(ADPKD)不同,常染色体隐性多囊肾病(ARPKD)一般不被认为与血管异常有关。此前仅报道过4例患有颅内动脉瘤的ARPKD患者。我们报告2例患有颅外血管异常的ARPKD患者:一名患有肾下主动脉和髂动脉瘤并伴有夹层的年轻男性,以及一名患有多个脾动脉和胃动脉瘤及动脉血管畸形的少女。这些病例引发了一个问题,即ARPKD中血管完整性和发育是否可能受损,或许是通过与ADPKD重叠的分子机制。小鼠研究支持了这种可能性,这些研究显示ARPKD基因在大血管壁中表达。