Law C J, Fisher A M, Temple I K
Wessex Regional Genetics Service, Southampton University Hospitals Trust, UK.
J Med Genet. 1998 Aug;35(8):685-9. doi: 10.1136/jmg.35.8.685.
We describe a 32 year old male with a distal 6p24.3-->pter deletion. He has specific developmental anomalies of the anterior chamber of the eye and a cleft uvula which is consistent with the recent localisation of genes for iris development and orofacial clefting to distal 6p. In addition he has progressive sensorineural deafness and this may localise a gene for deafness to this region. We conclude that a refined distal 6p deletion syndrome exists and includes a characteristic facial appearance with hypertelorism, downward slanting palpebral fissures, tented mouth, smooth philtrum, palatal malformation, ear anomalies, anterior chamber eye defects, progressive sensorineural deafness, cardiac defects, abdominal herniae, small external genitalia, and motor and speech delay.
我们描述了一名32岁男性,其存在6号染色体短臂24.3区至末端的缺失(6p24.3-->pter)。他有眼部前房的特定发育异常以及悬雍垂裂,这与近期将虹膜发育和口面部裂隙相关基因定位到6号染色体短臂末端的研究结果相符。此外,他还有进行性感觉神经性耳聋,这可能将一个耳聋相关基因定位到了该区域。我们得出结论,存在一种精细的6号染色体短臂末端缺失综合征,其特征包括面部外观异常,如眼距增宽、睑裂向下倾斜、口呈帐篷状、人中平滑、腭裂、耳部异常、眼前房缺陷、进行性感觉神经性耳聋、心脏缺陷、腹疝、外生殖器小以及运动和语言发育迟缓。