Hunter A G, Woerner S J, Montalvo-Hicks L D, Fowlow S B, Haslam R H, Metcalf P J, Lowry R B
Am J Med Genet. 1979;3(3):269-79. doi: 10.1002/ajmg.1320030305.
This paper describes six Hutterite children from five families who appear to have been affected by the same syndrome that was described in two brothers by Bowen and Conradi [1]. Our additional cases confirm that the major features of the syndrome include porportionate intrauterine growth retardation, microcephaly, micrognathia, a prominent nose, rocker-bottom feet, joint limitation, and failure to thrive, with death within the first year of life. Bowen-Conradi syndrome is an autosomal recessive trait and pedigree records show that all six families now known are related to each other through two couples born in the late 1700s but that there are additional earlier possible sources of the responsible gene. The differential diagnosis of this syndrome is discussed.
本文描述了来自五个家庭的六名哈特派儿童,他们似乎患有与鲍恩和康拉迪在两兄弟中所描述的相同综合征[1]。我们的其他病例证实,该综合征的主要特征包括匀称性宫内生长迟缓、小头畸形、小颌畸形、鼻子突出、摇椅底足、关节受限和发育不良,在出生后第一年内死亡。鲍恩-康拉迪综合征是一种常染色体隐性性状,家系记录显示,目前已知的所有六个家庭都通过18世纪末出生的两对夫妇相互关联,但可能还有更早的该致病基因来源。本文讨论了该综合征的鉴别诊断。