Varnero S, Becchi G, Bormida R, Martinengo E, Carozzi S
Servizio di Cardiologia, Ospedale San Paolo, Savona.
G Ital Cardiol. 1992 Jul;22(7):825-8.
The involvement of the cardiovascular system in hereditary connective tissue disorders has been known for a long time and produces abnormalities that usually affect valves and arterial walls. Valvular diseases in typical autosomal dominant illnesses such as polycystic kidney (PK) have recently been reported.
To test the prevalence of valvular prolapses in this disease, we studied [with clinical, chest x-ray, electrocardiographic, and echocardiographic (monobidimensional and Doppler) examination] 21 subjects with PK (Group A) and 34 unaffected relatives (Group B). 36 subjects comparable in sex, age, blood pressure, body build and renal function, with other kinds of kidney diseases, were the control group (Group C).
In group A, 7 subjects (33.3%) were found affected by mitral valve prolapse (MVP), of whom 3 also had mitral incompetence, 4 tricuspid valve prolapse and 2 aortic cusp prolapse with mild valvular insufficiency. Another 2 patients had evidence of mitral and aortic incompetence, respectively, without valvular prolapse, annulus ectasia or morphological features of a rheumatic valvular disease. In Group B, 8 subjects (23.5%) had MVP; In Group C, MVP was seen in only 1 patient (2.7%) and aortic incompetence in one other. Statistical analysis confirmed the significance of the greater prevalence of MVP in group A and B in comparison with group C (p < 0.01).
The high prevalence of valvular abnormalities in autosomal dominant PK suggests a common genetic disorder producing a defect in the extracellular matrix. An alternative hypothesis is that of two discrete yet adjacent genes.
心血管系统参与遗传性结缔组织疾病已为人所知甚久,且会产生通常影响瓣膜和动脉壁的异常情况。近期有报道称典型的常染色体显性疾病如多囊肾(PK)中存在瓣膜疾病。
为检测该疾病中瓣膜脱垂的患病率,我们[通过临床、胸部X线、心电图及超声心动图(一维和多普勒)检查]研究了21例PK患者(A组)和34名未患病亲属(B组)。36名在性别、年龄、血压、体型和肾功能方面与上述两组相当,但患有其他类型肾脏疾病的受试者作为对照组(C组)。
A组中,7名受试者(33.3%)被发现患有二尖瓣脱垂(MVP),其中3人同时存在二尖瓣关闭不全,4人有三尖瓣脱垂,2人有主动脉瓣叶脱垂伴轻度瓣膜关闭不全。另外2例患者分别有二尖瓣和主动脉瓣关闭不全的证据,但无瓣膜脱垂、瓣环扩张或风湿性瓣膜病的形态学特征。B组中,8名受试者(23.5%)患有MVP;C组中,仅1例患者(2.7%)有MVP,另有1例有主动脉瓣关闭不全。统计学分析证实,A组和B组中MVP患病率高于C组具有显著性意义(p < 0.01)。
常染色体显性PK中瓣膜异常的高患病率提示存在一种导致细胞外基质缺陷的常见遗传疾病。另一种假说是两个不连续但相邻的基因所致。