Richardson R J, Kirk J M
Hospital for Sick Children, London.
Arch Dis Child. 1990 Oct;65(10):1113-7. doi: 10.1136/adc.65.10.1113.
Eight children (four boys and four girls) with extreme failure to thrive, dysmorphic features, developmental delay, hypoparathyroidism, and abnormal skeletal survey were studied. They were the products of seven consanguinous marriages, two of the patients being brothers. In the remaining six families, a further four children had affected siblings who had died in infancy. When assessed the children were aged 0.47-12.8 years; SD scores were less than -2 for height, weight, and head circumference in all patients. The children had identical facies with deep set eyes, depressed nasal bridge with beaked nose, long philtrum, thin upper lip, micrognathia, and large floppy earlobes. They were all developmentally retarded. The following abnormalities were found on investigation: hypocalcaemia in all (of whom six of seven had hypoparathyroidism), medullary stenosis and other skeletal survey defects in seven of the eight children, and reduced numbers of T cell subsets in four of four tested. We believe that these children represent a new, as yet undescribed genetically determined syndrome.
对八名极度生长发育迟缓、存在畸形特征、发育延迟、甲状旁腺功能减退且骨骼检查异常的儿童(四名男孩和四名女孩)进行了研究。他们是七对近亲婚姻的后代,其中两名患者是兄弟。在其余六个家庭中,还有另外四名儿童有患病的兄弟姐妹,这些患病的兄弟姐妹在婴儿期就已死亡。评估时,这些儿童的年龄在0.47至12.8岁之间;所有患者的身高、体重和头围标准差得分均低于 -2。这些儿童面部特征相同,眼睛深陷,鼻梁凹陷呈鹰嘴鼻,人中长,上唇薄,小颌畸形,耳垂大且松软。他们均存在发育迟缓。检查发现以下异常情况:所有人均有低钙血症(其中七人中有六人患有甲状旁腺功能减退),八名儿童中有七人存在髓腔狭窄和其他骨骼检查缺陷,四名接受检测的儿童中有四人T细胞亚群数量减少。我们认为这些儿童代表了一种新的、尚未被描述的基因决定综合征。