Houston C S, Shokeir M H
J Can Assoc Radiol. 1981 Dec;32(4):215-9.
The Pena-Shokeir I syndrome is characterized by prenatal onset of growth deficiency, a specific constellation of facial features, multiple ankyloses, camptodactyly, and talipes equinovarus and is almost invariably fatal. A common and perhaps specific radiographic sign is that of subluxations at interphalangeal joints of the fingers, present in four of six patients described here. This autosomal recessive syndrome should be differentiated from arthrogryposis multiplex congenita on the basis of the unusual facial configuration and early lethality, even when interphalangeal subluxations are absent.
佩纳-绍凯尔I型综合征的特征为产前生长发育迟缓、特定的面部特征组合、多处关节强直、屈曲指及马蹄内翻足,且几乎无一例外是致命的。一个常见且可能具有特异性的影像学表现是手指指间关节半脱位,在此描述的6例患者中有4例出现此表现。即使不存在指间关节半脱位,基于其不同寻常的面部形态和早期致死性,这种常染色体隐性遗传综合征也应与先天性多发性关节挛缩症相鉴别。