Pees C, Michel-Behnke I, Hagl M, Laccone F
Pediatric Marfan Syndrome Outpatient Clinic, Medical University Vienna, Vienna, Austria; Department of Pediatric Cardiology, Pediatric Heart Center Vienna, University Children's Hospital, Vienna, Austria.
Clin Genet. 2014 Dec;86(6):552-7. doi: 10.1111/cge.12314. Epub 2013 Dec 4.
We report about 52 pediatric patients of 40 different families with confirmed Marfan syndrome (MFS) in 49 patients and Loeys-Dietz syndrome (LDS) in 3 patients. We found 39 different mutations, 15 of them being novel. Phenotype-genotype correlation in the 49 MFS patients showed that the majority of patients carrying mutations in exons 1-21 had ectopic lens (80%). Patients having mutations in exons 23-32 had a higher probability of aortic root dilation, in 50% even above a z score of 3. We found three children with neonatal MFS form, two of them with novel mutations. Of the three LDS patients, only one presented with the typical phenotype of LDS type 1.
我们报告了来自40个不同家庭的52例儿科患者,其中49例确诊为马凡综合征(MFS),3例确诊为洛伊迪茨综合征(LDS)。我们发现了39种不同的突变,其中15种是新发现的。49例MFS患者的表型-基因型相关性显示,大多数在外显子1-21携带突变的患者有晶状体异位(80%)。在外显子23-32携带突变的患者主动脉根部扩张的可能性更高,50%的患者甚至z评分高于3。我们发现3名患有新生儿型MFS的儿童,其中2名有新的突变。在3例LDS患者中,只有1例表现出典型的1型LDS表型。