Niikawa N, Fukushima Y, Taniguchi N, Iizuka S, Kajii T
Hum Genet. 1982;60(4):373-5. doi: 10.1007/BF00569223.
Two unrelated patients with clinical features of 11p13 deletion syndrome, 3 years old and 3 months old, are reported. The clinical features of the patients included mental retardation, aniridia, nystagmus, blepharophimosis, and genitourinary abnormalities. Both patients were apparently free from Wilms' tumor and gonadoblastoma. Prometaphase banding analyses revealed a 46,XY,del(11)(p1300p1500) karyotype in one patient and 46,XX,dir ins(11;2)(p13;q12q23) in the other. Catalase activities in the erythrocytes in the two patients were respectively 65% and 56% of those of normal controls, close to the expected values in hemizygosity of the catalase gene. These findings confirmed a close linkage of the gene for catalase and those for the aniridia--Wilm's tumor or gonadoblastoma complex.
报告了两名患有11p13缺失综合征临床特征的无关患者,年龄分别为3岁和3个月。患者的临床特征包括智力发育迟缓、无虹膜、眼球震颤、睑裂狭小和泌尿生殖系统异常。两名患者显然均未患威尔姆斯瘤和性腺母细胞瘤。前中期显带分析显示,一名患者的核型为46,XY,del(11)(p1300p1500),另一名患者的核型为46,XX,dir ins(11;2)(p13;q12q23)。两名患者红细胞中的过氧化氢酶活性分别为正常对照的65%和56%,接近过氧化氢酶基因半合子状态下的预期值。这些发现证实了过氧化氢酶基因与无虹膜-威尔姆斯瘤或性腺母细胞瘤复合体相关基因之间存在紧密联系。