Dallapiccola B, Bricarelli F D, Quartino A R, Mazzilli M C, Chisci R, Gandini E
Acta Genet Med Gemellol (Roma). 1978;27:57-66. doi: 10.1017/s000156600000951x.
Two unrelated patients carrying imbalances involving the long arm of chromosome 6 are described. In the first trisomy 6q21 leads to qter had segregated from a maternal translocation t(6;16)(q15;q24). The clinical data of the proposita are compared with those of three other published cases. A partial 6q trisomy syndrome is postulated characterized by: growth deficiency of prenatal onset, psychomotor retardation, craniofacial abnormalities (microcephalia, hypertelorism, downward slanting palpebral fissures, flattened nasal bridge, long philtrum, hypoplastic perioral features, large jaw resulting in a round appearance of the face, receding chin, malformed ears) and dysmorphic extremities (contractures of limbs due to short flexor tendons, hypoplastic fingers, toes and nails). In the second case, monosomy 6q221 leads to qter resulted from a de novo rearrangement and was responsible for mental retardation and facial dysmorphism (reduced biparietal diameter, hypotelorism, absent eyebrows, prominent nose, ptosis, receding chin, dysmorphic ears). Studies of HLA and PGM3 segregation showed normal inheritance patterns and ruled out the location of these genes in bands 6q221 leads to qter.
本文描述了两名携带涉及6号染色体长臂失衡的无关患者。在第一例中,6q21至qter的三体性从母亲的t(6;16)(q15;q24)易位中分离出来。将先证者的临床数据与其他三例已发表病例的数据进行了比较。推测部分6q三体综合征的特征为:产前起病的生长发育迟缓、精神运动发育迟缓、颅面畸形(小头畸形、眼距增宽、睑裂向下倾斜、鼻梁扁平、人中长、口周特征发育不全、大下巴导致面部呈圆形、下巴后缩、耳朵畸形)和肢体畸形(由于屈肌腱短导致肢体挛缩、手指、脚趾和指甲发育不全)。在第二例中,6q221至qter的单体性是由新发重排导致的,导致智力发育迟缓和面畸形(双顶径减小、眼距过窄、无眉毛、鼻梁突出、上睑下垂、下巴后缩、耳朵畸形)。HLA和PGM3分离研究显示正常的遗传模式,并排除了这些基因位于6q221至qter区域的可能性。