Vernant P, Corone P, Rossignol A M, Bielman C
Arch Mal Coeur Vaiss. 1980 Jun;73(6):661-6.
120 cases of the Williams-Beuren syndrome were collected by 11 cardiological centres in France, to determine the incidence of the various clinical signs and to obtain information on its aetiology, outcome and treatment. The selection criteria for inclusion in the series was typical facies. No particular complications were reported during pregnancy. Boys were a little more affected than girls. The birth weight was low and problems, especially digestive, often occurred in the first months. Cardiac signs were usually detected from the first year, although the exact diagnosis was usually made later on. 3/4 patients had subaortic stenosis, which was severe in 1/3 cases. Involvement of the branches of the aorta was not looked for systematically: the incidence (1/5 cases) found was lower than the true figure. Half the patients also had stenoses on branches of the pulmonary artery, but only rarely were they severe. These vascular malformations often seem to be progressive and, over a 10 year period, half the patients deteriorated. Many extracardiac abnormalities were reported. The most frequently encountered were inguinal and/or umbilical hernia. Surgery on the subvalvular stenosis gave good results in over 80 p. 100 cases; operative mortality was about 10 p. 100. Surgery should be performed before irreversible coronary or myocardial lesions develop. The study of the previous history did not give any new information on the cause of the syndrome, whose association with idiopathic hypercalcaemia of infancy is emphasised once again.
法国11个心脏病中心收集了120例威廉姆斯-贝伦综合征病例,以确定各种临床症状的发生率,并获取有关其病因、预后和治疗的信息。该系列纳入标准为典型面容。孕期未报告特殊并发症。男孩受累略多于女孩。出生体重低,尤其是消化问题常在头几个月出现。心脏体征通常在第一年即可检测到,不过确切诊断通常在之后做出。3/4的患者有主动脉瓣下狭窄,其中1/3病例狭窄严重。未系统检查主动脉分支受累情况:所发现的发生率(1/5病例)低于实际数字。一半患者肺动脉分支也有狭窄,但严重的情况很少见。这些血管畸形似乎常呈进行性发展,在10年期间,一半患者病情恶化。报告了许多心脏外异常情况。最常见的是腹股沟和/或脐疝。瓣下狭窄手术在超过80%的病例中效果良好;手术死亡率约为10%。应在不可逆的冠状动脉或心肌病变出现之前进行手术。对既往病史的研究未提供关于该综合征病因的任何新信息,再次强调了其与婴儿特发性高钙血症的关联。