Hou J W, Wang T R
Department of Paediatrics, National Taiwan University Hospital, Taipei.
J Med Genet. 1996 Oct;33(10):879-81. doi: 10.1136/jmg.33.10.879.
We report a female baby with multiple congenital anomalies including left upper amelia, congenital short bowel with malrotation and pseudo-obstruction, dextrocardia with situs solitus, patent ductus arteriosus, and a tiny atrophic spleen. Chromosome study showed de novo 46,XX/46,XX,-4, + r(4)(p16-->q22.3)/47,XX,4, + r(4) (p16-->q22.3), + del(4)(pter-->q22.3:). The clinical findings in the patient were probably caused by the interaction of partial trisomy 4pter-->q22.3 or 4p16-->q22.3 and partial monosomy of 4q22.3-->4qter. This karyotype and phenotype have not previously been reported.
我们报告了一名患有多种先天性异常的女婴,包括左上臂缺如、先天性短肠伴旋转不良和假性肠梗阻、右位心伴内脏正位、动脉导管未闭以及微小萎缩脾脏。染色体研究显示为新发的46,XX/46,XX,-4, + r(4)(p16→q22.3)/47,XX,4, + r(4) (p16→q22.3), + del(4)(pter→q22.3:)。患者的临床发现可能是由4号染色体pter→q22.3或4p16→q22.3部分三体与4q22.3→4qter部分单体的相互作用引起的。此前尚未报道过这种核型和表型。