Wallerstein R, Scott C I, Nicholson L
Division of Medical Genetics, Alfred I. DuPont Institute, Wilmington, Delaware, USA.
Am J Med Genet. 1997 Jun 13;70(3):267-72. doi: 10.1002/(sici)1096-8628(19970613)70:3<267::aid-ajmg10>3.0.co;2-j.
We present a boy followed from age 5-13 years who is the fifth reported case of ter Haar syndrome. This is a recently-named entity comprising congenital glaucoma, hypertelorism, congenital heart defects and kyphoscoliosis, skeletal dysplasia, and developmental delay. These patients were originally thought to have an autosomal-recessive form of Melnick-Needles syndrome, and were only identified as having a distinct syndrome with the report of the fourth case. Probable autosomal-recessive inheritance is based on consanguinity in 4 of 5 cases. Ocular, cardiac, and craniofacial findings distinguish ter Haar syndrome as a distinct entity. Our patient is the longest survivor at present, suggesting that there is heterogeneity in this syndrome or, alternatively, that aggressive therapy of the congenital heart defects has significant effect.
我们报告一名从5岁至13岁进行随访的男孩,他是第五例被报道的特拉尔综合征患者。这是一个最近命名的疾病实体,包括先天性青光眼、眼距增宽、先天性心脏缺陷、脊柱后凸侧弯、骨骼发育异常和发育迟缓。这些患者最初被认为患有常染色体隐性形式的梅尼克-尼德尔斯综合征,直到第四例病例报告后才被确定为患有独特的综合征。5例中有4例存在近亲结婚,提示可能为常染色体隐性遗传。眼部、心脏和颅面部表现使特拉尔综合征成为一个独特的疾病实体。我们的患者是目前存活时间最长的,这表明该综合征存在异质性,或者说,对先天性心脏缺陷的积极治疗具有显著效果。