Bonnet D, Cormier-Daire V, Kachaner J, Szezepanski I, Souillard P, Sidi D, Munnich A, Lyonnet S
Unité de Recherches sur les Handicaps Génétiques de l'Enfant-INSERM U-393, Hôpital des Enfants Malades, Paris, France.
Am J Med Genet. 1997 Jan 20;68(2):182-4.
Cono-truncal cardiac malformations account for some 50% of congenital heart defects in newborn infants. Recently, hemizygosity for chromosome 22q11.2 was reported in patients with the DiGeorge/Velo-cardio-facial syndromes (DGS/VCFS) and causally related disorders. We have explored the potential use of microsatellite DNA markers for rapid detection of 22q11 deletions in 19 newborn infants referred for cono-truncal heart malformations with associated DGS/VCFS anomalies. A failure of parental inheritance was documented in 84.2% of cases (16/19). PCR-based genotyping using microsatellite DNA markers located within the commonly deleted region allowed us either to confirm or reject a 22q11 microdeletion in 94.3% of cases (18/19) within 24 hours. This test is now currently performed in the infants referred to us for a cono-truncal heart malformation as a first intention screening for 22q11 microdeletion.
圆锥干型心脏畸形约占新生儿先天性心脏缺陷的50%。最近,据报道,患有DiGeorge/心脏颜面综合征(DGS/VCFS)及相关疾病的患者存在22号染色体q11.2半合子状态。我们研究了微卫星DNA标记物在快速检测19例因圆锥干型心脏畸形伴发DGS/VCFS异常而转诊的新生儿22q11缺失方面的潜在用途。84.2%的病例(16/19)记录到亲代遗传缺失。使用位于常见缺失区域内的微卫星DNA标记物进行基于聚合酶链反应(PCR)的基因分型,使我们能够在24小时内对94.3%的病例(18/19)确认或排除22q11微缺失。目前,对于因圆锥干型心脏畸形转诊至我们这里的婴儿,该检测作为22q11微缺失的首选筛查方法正在进行。