Claeys I, Holvoet M, Eyskens B, Adriaensens P, Gewillig M, Fryns J P, Devriendt K
Department of Pediatrics, University Hospital Leuven, Belgium.
Am J Med Genet. 1997 Sep 19;74(5):515-20. doi: 10.1002/(sici)1096-8628(19970919)74:5<515::aid-ajmg12>3.0.co;2-f.
We report the clinical findings in 5 patients with a terminal deletion of the short arm of chromosome 8. Mild developmental delay was constantly present, in association with microcephaly in 4 of 5 patients. Facial anomalies were mild or absent. A congenital heart defect was present in 3 patients: an atrioventricular septal defect (AVSD) in 2 and an atrial septal defect type II (ASDII) with pulmonary stenosis in one. A highly similar pattern of behavioural difficulties was present in the 3 older children (8-11 years), with outbursts of aggressiveness and destructive behaviour. Follow-up in one patient showed that at the age of 16 years, these behavioural problems had largely disappeared. This observation suggests that in addition to mental retardation, microcephaly, congenital heart defect (typically AVSD), a terminal deletion of chromosome 8p may be associated with a characteristic behavioural phenotype during childhood.
我们报告了5例8号染色体短臂末端缺失患者的临床发现。5例患者中有4例存在轻度发育迟缓,并伴有小头畸形。面部异常轻微或无异常。3例患者存在先天性心脏缺陷:2例为房室间隔缺损(AVSD),1例为伴有肺动脉狭窄的II型房间隔缺损(ASDII)。3名年龄较大的儿童(8 - 11岁)表现出高度相似的行为困难模式,有攻击性和破坏性行为发作。对1例患者的随访显示,在16岁时,这些行为问题已基本消失。这一观察结果表明,除智力迟钝、小头畸形、先天性心脏缺陷(典型的AVSD)外,8号染色体短臂末端缺失可能与儿童期特征性的行为表型有关。