Teo S H, Chan D K, Yong M H, Ng I S, Wong K Y, Knight L, Ho L Y
Department of Paediatric Medicine, Singapore General Hospital, Singapore.
Ann Acad Med Singap. 1997 May;26(3):360-4.
Williams syndrome first described in 1961 is generally characterised by mental deficiency, gregarious personality, unusual "elfin" facies, supravalvular aortic stenosis and idiopathic infantile hypercalcaemia. Patients with Williams syndrome show a hemizygous submicroscopic deletion of 7q11.23 detectable by fluorescent in-situ hybridisation (FISH). The deleted portion of the chromosome corresponds to the Elastin gene. We report 3 girls with characteristics of Williams syndrome in whom the diagnosis was confirmed by demonstration of the hemizygous deletion of 7q11.23 in the karyotype by FISH. These patients, aged 6, 7 and 10 years, showed the characteristic facies and gregarious personalities. Some developmental delay with mild mental deficiency and dysmorphic facies were prominent features in the initial presentation. Cardiac lesions found in these patients were small patent ductus arteriosus which closed, pulmonary valvular stenosis and mitral valve prolapse associated with mitral regurgitation respectively. Hypercalcaemia was not documented in these patients. Learning difficulty was a major issue and all patients required special schooling. Chromosome analyses done on peripheral blood were found to be normal in all patients. FISH using the Elastin Williams Syndrome Chromosome Region (WSCR) probes (oncor) showed the hemizygous deletion of 7q11.23. Diagnosis of Williams syndrome can now be confidently confirmed with the help of FISH.
威廉姆斯综合征于1961年首次被描述,其一般特征为智力缺陷、性格合群、特殊的“小精灵样”面容、主动脉瓣上狭窄和特发性婴儿高钙血症。威廉姆斯综合征患者显示7q11.23存在半合子亚显微缺失,可通过荧光原位杂交(FISH)检测到。染色体的缺失部分对应于弹性蛋白基因。我们报告了3名具有威廉姆斯综合征特征的女孩,通过FISH证实其核型中存在7q11.23半合子缺失,从而确诊。这些患者年龄分别为6岁、7岁和10岁,表现出特征性面容和合群的性格。最初表现的突出特征为一些发育迟缓、轻度智力缺陷和畸形面容。这些患者中发现的心脏病变分别为已闭合的小型动脉导管未闭、肺动脉瓣狭窄和伴有二尖瓣反流的二尖瓣脱垂。这些患者未记录到高钙血症。学习困难是一个主要问题,所有患者都需要特殊教育。所有患者外周血染色体分析均正常。使用弹性蛋白威廉姆斯综合征染色体区域(WSCR)探针(oncor)进行的FISH显示7q11.23半合子缺失。借助FISH现在可以可靠地确诊威廉姆斯综合征。