Habedank M, Trost-Brinkhues G
J Med Genet. 1983 Oct;20(5):377-9. doi: 10.1136/jmg.20.5.377.
A three generation pedigree is described in which there are two carriers of translocation t(7;18). Two members of the family have trisomy 18p and a stillborn child had monosomy 18p and holoprosencephaly. Another stillborn child probably had holoprosencephaly; the karyotype was not analysed. Based on this observation, the occasional occurrence of holoprosencephaly in monosomy 18p (10% of previously reported cases) may not be the result of the expression of a recessive mutant gene in the hemizygous state, as assumed up to now.
本文描述了一个三代家系,其中有两名7号和18号染色体易位携带者。该家族中有两名成员患有18号染色体短臂三体,一名死产儿患有18号染色体短臂单体及前脑无裂畸形。另一名死产儿可能患有前脑无裂畸形;未对其核型进行分析。基于这一观察结果,18号染色体短臂单体中偶尔出现的前脑无裂畸形(占既往报道病例的10%)可能并非如目前所认为的那样,是半合子状态下隐性突变基因表达的结果。