Kim Jeong Hyun, Oh Phil Soo, Na Hye Yeon, Kim Sun-Hee, Cho Hyoun Chan
Department of Laboratory Medicine, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.
Korean J Lab Med. 2009 Feb;29(1):77-81. doi: 10.3343/kjlm.2009.29.1.77.
Ring chromosome is a structural abnormality that is thought to be the result of fusion and breakage in the short and long arms of chromosome. Wolf-Hirschhorn syndrome (WHS) is a well-known congenital anomaly in the ring chromosome 4 with a partial deletion of the distal short arm. Here we report a 10-month-old male of mosaic ring chromosome 4 with the chief complaint of severe short stature. He showed the height of -4 standard deviation, subtle hypothyroidism and mild atrial septal defect/ventricular septal defect, and also a mild language developmental delay was suspected. Brain magnetic resonance imaging showed multifocal leukomalacia. Chromosomal analysis of the peripheral blood showed the mosaic karyotype with [46,XY,r(4)(p16q35)[84]/45,XY,-4[9]/91,XXYY, dic r(4;4)(p16q35;p16q35)[5]/46,XY,dic r(4;4)(p16q35;p16q35)[2]]. FISH study showed the deletion of the 4p subtelomeric region with the intact 4q subtelomeric and WHS region. Both paternal and maternal karyotypes were normal. We compared the phenotypic variation with the previously reported cases of ring chromosome 4. The ring chromosome 4 with the subtelomeric deletion of short arm seems to be related with the phenotype of short stature.
环状染色体是一种结构异常,被认为是染色体短臂和长臂融合及断裂的结果。沃尔夫-赫希霍恩综合征(WHS)是环状染色体4上一种著名的先天性异常,伴有远端短臂部分缺失。在此,我们报告一名10个月大的患有嵌合型环状染色体4的男性,主要症状为严重身材矮小。他的身高低于标准值4个标准差,存在轻微甲状腺功能减退和轻度房间隔缺损/室间隔缺损,并且还疑似存在轻度语言发育迟缓。脑部磁共振成像显示多灶性脑白质软化。外周血染色体分析显示嵌合核型为[46,XY,r(4)(p16q35)[84]/45,XY,-4[9]/91,XXYY, dic r(4;4)(p16q35;p16q35)[5]/46,XY,dic r(4;4)(p16q35;p16q35)[2]]。荧光原位杂交研究显示4p亚端粒区域缺失,而4q亚端粒和WHS区域完整。父母双方的核型均正常。我们将该病例的表型变异与先前报道的环状染色体4病例进行了比较。短臂亚端粒缺失的环状染色体4似乎与身材矮小的表型有关。