Lin A E, Doshi N, Flom L, Tenenholz B, Filkins K L
Department of Medical Genetics, Western Pennsylvania Hospital, Pittsburgh.
Am J Med Genet. 1991 Jun 1;39(3):247-51. doi: 10.1002/ajmg.1320390303.
We report on an infant girl with hydrops, macrocephaly, high forehead, flat face, hypertelorism, broad nasal bridge, median cleft lip and alveolar ridge, grooved palate, accessory frenula, small tongue, milia, severe rib and limb shortness, brachydactyly, talipes equinovarus, Dandy-Walker malformation, accessory spleen, unfixed mesentery, ectopic pancreas, and renal cysts. This patient resembles seven previously reported patients with the Beemer-Langer syndrome, a distinct lethal short rib syndrome characterized by hydrops, markedly short ribs and limbs, median cleft lip with or without cleft palate, flat face, and macrocephaly. Polydactyly is usually absent. Our patient's oral anomalies suggest an orofaciodigital syndrome, but the severe rib and limb shortness distinguish it from those disorders.
我们报告了一名患有水肿、巨头畸形、高额、扁平脸、眼距过宽、鼻梁宽、唇腭裂和牙槽嵴裂、腭裂、副系带、小舌、粟丘疹、严重肋骨和肢体短小、短指畸形、马蹄内翻足、Dandy-Walker畸形、副脾、肠系膜未固定、异位胰腺和肾囊肿的女婴。该患者与之前报道的7例Beemer-Langer综合征患者相似,这是一种独特的致死性短肋综合征,其特征为水肿、肋骨和肢体明显短小、有或无腭裂的唇裂、扁平脸和巨头畸形。通常无多指畸形。我们患者的口腔异常提示口面指综合征,但严重的肋骨和肢体短小使其与这些疾病相区别。