Chen Chih-Ping, Lin Shuan-Pei, Chern Schu-Rern, Wu Peih-Shan, Chang Shuenn-Dyh, Ng Shu-Hang, Liu Yu-Peng, Su Jun-Wei, Wang Wayseen
Department of Medicine, Mackay Medical College, New Taipei City, Taiwan.
Eur J Med Genet. 2012 Nov;55(11):666-9. doi: 10.1016/j.ejmg.2012.07.003. Epub 2012 Jul 25.
We report a 26-month-old girl with profound hearing impairment, microcephaly, psychomotor retardation, short palpebral fissures, hypertelorism, epicanthic folds, a broad nasal bridge, anteverted nostrils, large low-set ears, micrognathia, brachymesophalangy of the second and the fifth fingers, clinodactyly of bilateral fifth fingers and a wide interdigital space between the first and the second toes, carrying a 4.4-Mb de novo microdeletion of chromosome 2p24.3 → p24.2. This region contains the genes of FAM84A, NBAS, DDX1, MYCNOS and MYCN, of which haploinsufficiency or mutations of the MYCN gene is associated with Feingold syndrome. Brain magnetic resonance imaging revealed cochlear nerve hypoplasia and internal auditory canal stenosis. Temporal bone computed tomography showed abnormal dilation of bilateral vestibular and lateral semicircular canals. The present case provides evidence that haploinsufficiency of MYCN in 2p24.3 deletion in humans can cause structural and functional abnormalities of the inner ear.
我们报告了一名26个月大的女童,她患有严重听力障碍、小头畸形、精神运动发育迟缓、睑裂短小、眼距增宽、内眦赘皮、鼻梁宽、鼻孔前倾、低位大耳、小颌畸形、第二和第五指中节指骨短小、双侧第五指弯曲指畸形以及第一和第二趾间间隙增宽,其携带2号染色体2p24.3→p24.2处4.4Mb的新发微缺失。该区域包含FAM84A、NBAS、DDX1、MYCNOS和MYCN基因,其中MYCN基因的单倍剂量不足或突变与费因戈尔德综合征相关。脑磁共振成像显示蜗神经发育不全和内耳道狭窄。颞骨计算机断层扫描显示双侧前庭和外侧半规管异常扩张。本病例提供了证据,表明人类2p24.3缺失中MYCN的单倍剂量不足可导致内耳的结构和功能异常。