Mikhail Fady M, McIlvried Dawn, Holt R Lynn, Messiaen Ludwine, Descartes Maria D, Carroll Andrew J
Department of Genetics, School of Medicine, University of Alabama at Birmingham, Birmingham, Alabama 35294, USA.
Am J Med Genet A. 2006 Aug 1;140(15):1647-54. doi: 10.1002/ajmg.a.31330.
We report on an 8-year-old girl with near-complete trisomy 17p syndrome due to a de novo unbalanced t(14;17)(p11.2;p11.2). She has features consistent with the previously described cases with complete trisomy 17p, including pre- and post-natal growth retardation, motor and mental retardation, skeletal anomalies, clinodactyly of the 5th finger, hypertrichosis, as well as facial characteristics including microcephaly, receding forehead, ptosis, low-set malformed ears, smooth philtrum, high-arched palate, and a short broad neck. Fluorescence in situ hybridization showed that the breakpoints were p11.2 for both chromosome 14 and 17. Microsatellite analysis showed that the duplicated 17p was of paternal origin, and indicated that the breakpoint involving 17p11.2 is most likely located within the approximately 1-Mb segment from the centromere, and not involving the proximal Smith-Magenis syndrome (SMS) low copy repeat. We compare the clinical features of our patient with those previously reported to further delineate the phenotype of complete trisomy 17p syndrome.
我们报告了一名8岁女孩,因新发不平衡易位t(14;17)(p11.2;p11.2)导致近乎完全的17p三体综合征。她具有与先前报道的完全17p三体病例一致的特征,包括产前和产后生长发育迟缓、运动和智力发育迟缓、骨骼异常、第5指斜指畸形、多毛症,以及面部特征,包括小头畸形、前额后缩、上睑下垂、低位畸形耳、人中平滑、高拱腭和短而宽的颈部。荧光原位杂交显示14号和17号染色体的断点均为p11.2。微卫星分析表明,重复的17p来自父系,并且表明涉及17p11.2的断点最有可能位于距着丝粒约1 Mb的片段内,且不涉及近端史密斯-马吉尼斯综合征(SMS)低拷贝重复序列。我们将我们患者的临床特征与先前报道的特征进行比较,以进一步描绘完全17p三体综合征的表型。