Cazorla Calleja María R, Verdú Alfonso, Félix Valentin
Neuropediatric Unit, Hospital Virgen de la Salud, Avenida de Barber 30, 45004 Toledo, Spain.
Brain Dev. 2003 Apr;25(3):220-3. doi: 10.1016/s0387-7604(02)00211-5.
An infant with Dandy-Walker malformation and prenatally diagnosed tetrasomy 9p is reported. Chromosomal analysis of primary amniocyte culture revealed true mosaicism for two cell lines: 50% of the cells had an isochromosome 9p (pter-q13::q13-pter), and the other 50% showed a normal female karyotype (46,XX). After birth the same chromosomal abnormality was found in 75% of peripheral blood lymphocytes. Phenotypic features included intrauterine growth retardation, hypotrophy of the left side of the body with left microphthalmus, and progressive hydrocephalus secondary to Dandy-Walker malformation. Although most cases of Dandy-Walker malformation are not associated with chromosomal abnormalities, our case, together with two previously reported cases of the same association, indicates that this chromosomal disorder should be looked for in children with Dandy-Walker malformation and abnormal somatic development.
报告了一名患有丹迪-沃克畸形且产前诊断为9号染色体短臂四体的婴儿。对原代羊膜细胞培养物进行染色体分析发现了两种细胞系的真正嵌合体:50%的细胞具有9号等臂染色体(pter-q13::q13-pter),另外50%显示正常女性核型(46,XX)。出生后,在外周血淋巴细胞中发现相同的染色体异常的比例为75%。表型特征包括宫内生长迟缓、身体左侧发育不全伴左侧小眼畸形,以及继发于丹迪-沃克畸形的进行性脑积水。虽然大多数丹迪-沃克畸形病例与染色体异常无关,但我们的病例以及之前报道的另外两例相同关联的病例表明,对于患有丹迪-沃克畸形和躯体发育异常的儿童应进行这种染色体疾病的检查。