Hedera Peter, Innis Jeffrey W
Department of Pediatrics, Division of Genetics, University of Michigan, 1500 E. Medical Center Drive, Ann Arbor, MI 48109-0618, USA.
Am J Med Genet A. 2003 Oct 15;122A(3):257-60. doi: 10.1002/ajmg.a.20263.
We report a patient with severe mental retardation (MR), microcephaly, Dandy-Walker malformation, bilateral lip/palate clefts, hypertrophied sublingual frenulum, lobular tongue, absent thumbs, and other skeletal abnormalities, including Y-shaped metacarpals and urogenital abnormalities. High-resolution karyotype and subtelomeric fluorescence in situ hybridization were normal. We propose that his clinical picture is most consistent with Juberg-Hayward or orocraniodigital syndrome. Several clinical features present in our patient (unilateral distal displacement of elbow position, second-site radioulnar synostosis, bilateral Y-shaped metacarpal, lobular tongue, hypertrophic frenuli, Dandy-Walker malformation) have not previously been reported in this condition, thus expanding the phenotypic spectrum of this rare condition. The presence of these novel findings suggests possible overlap with other syndromes, such as orofaciodigital and Malpuech syndromes.
我们报告了一名患有严重智力障碍(MR)、小头畸形、丹迪-沃克畸形、双侧唇腭裂、舌下系带肥厚、小叶状舌、拇指缺如以及其他骨骼异常(包括Y形掌骨)和泌尿生殖系统异常的患者。高分辨率核型分析和亚端粒荧光原位杂交结果均正常。我们认为他的临床表现与朱伯格-海沃德综合征或口颅指综合征最为相符。我们的患者所具有的一些临床特征(肘部位置单侧远端移位、第二部位桡尺骨融合、双侧Y形掌骨、小叶状舌、系带肥厚、丹迪-沃克畸形)此前在该病症中尚未有报道,从而扩展了这种罕见病症的表型谱。这些新发现的存在提示可能与其他综合征存在重叠,如口面指综合征和马尔皮克综合征。