Gelman-Kohan Z, Antonelli J, Ankori-Cohen H, Adar H, Chemke J
Clinical Genetics Unit, Kaplan Hospital, Rehovot, Israel.
Eur J Pediatr. 1991 Sep;150(11):797-9. doi: 10.1007/BF02026715.
The acrocallosal syndrome (ACS) was recognized by Schinzel in 1979 as a specific entity, characterized by the association of craniofacial anomalies, total or partial agenesis of corpus callosum, polysyndactyly and mental retardation. The inheritance is autosomal recessive, based on instances of recurrence in siblings and cousins and parental consanguinity. A large inbred kindred with recurrent ACS is presented. This family further strengthens the hypothesis of autosomal recessive inheritance for this syndrome. The array of clinical manifestations in this sibship and those previously reported exemplify the phenomenon of inter- and intrafamilial variability that must be considered when defining ACS. Based on a review of published reports and the present family, essential, additional and occasional findings are distinguished. Attention is drawn to geographical clustering of the families.
1979年,施因策尔将顶枕连合综合征(ACS)确认为一种特殊病症,其特征为颅面畸形、胼胝体完全或部分发育不全、多指(趾)畸形以及智力发育迟缓。基于兄弟姐妹和表亲的复发实例以及父母近亲结婚情况,该病症为常染色体隐性遗传。本文展示了一个患有复发性ACS的大型近亲家族。这个家族进一步强化了该综合征常染色体隐性遗传的假说。该家族以及先前报道的家族中一系列临床表现例证了在定义ACS时必须考虑的家族间和家族内变异性现象。基于对已发表报告和当前家族情况的回顾,区分了主要、附加和偶然发现。文中还提到了这些家族的地理聚集情况。