Narahara K, Kikkawa K, Murakami M, Hiramoto K, Namba H, Tsuji K, Yokoyama Y, Kimoto H
Department of Pediatrics, Okayama University School of Medicine, Japan.
Am J Med Genet. 1990 Feb;35(2):269-73. doi: 10.1002/ajmg.1320350225.
Two patients with monosomy for the distal portion of the short arm of chromosome 3 are described. Chromosome analysis on prometaphase cells demonstrated a karyotype of 46,XX,del(3) (p25.3) in one patient and 46,XX,r(3)(p26.1q29) in the other. The former patient showed characteristic clinical manifestations of the 3p- syndrome, including growth failure, mental retardation, microcephaly with a flat occiput, triangular face, synophrys, blepharoptosis, hypertelorism, broad and flat nose, long philtrum, down-turned mouth, micrognathia, apparently lowset and malformed ears, fingers abnormalities, and deafness. The latter patient had a nonspecific phenotype with mental retardation, growth failure and microcephaly. Karyotype-phenotype comparisons in the present cases and 16 previously reported cases with deficiency of the distal portion of 3p suggests that deficiency of the 3p25.3 band is critical to produce the main clinical manifestations of the del(3p) syndrome.
本文描述了两名3号染色体短臂远端单体型患者。对前中期细胞进行的染色体分析显示,一名患者的核型为46,XX,del(3)(p25.3),另一名患者的核型为46,XX,r(3)(p26.1q29)。前一名患者表现出3p-综合征的典型临床表现,包括生长发育迟缓、智力障碍、枕部扁平的小头畸形、三角脸、连眉、上睑下垂、眼距增宽、宽扁鼻、长人中、嘴角下垂、小颌畸形、耳朵明显低位且畸形、手指异常和耳聋。后一名患者具有非特异性表型,表现为智力障碍、生长发育迟缓和小头畸形。对本病例以及之前报道的16例3p远端缺失病例进行的核型-表型比较表明,3p25.3带的缺失对于产生del(3p)综合征的主要临床表现至关重要。