Merrer M L, Briard M L, Girard S, Mulliez N, Moraine C, Imbert M C
Clinique et Unité de Recherches de Génétique Médicale INSERM U.12, Hôpital des Enfants Malades, Paris, France.
J Med Genet. 1988 Feb;25(2):88-95. doi: 10.1136/jmg.25.2.88.
We report eight cases of a lethal association of failure to thrive, facial dysmorphism, ambiguous genitalia, syndactyly, postaxial polydactyly, and internal developmental anomalies (Hirschsprung's disease, cardiac and renal malformation). This syndrome is likely to be autosomal recessive and resembles Smith-Lemli-Opitz (SLO) syndrome. However, the lethality, the common occurrence of polydactyly, and the sexual ambiguity distinguishes this condition from SLO syndrome. A review of published reports supports the separate classification of this syndrome for which we propose the name lethal acrodysgenital dwarfism.
我们报告了8例病例,这些病例存在生长发育迟缓、面部畸形、生殖器模糊、并指、轴后多指以及内部发育异常(先天性巨结肠、心脏和肾脏畸形)的致命关联。这种综合征可能为常染色体隐性遗传,与史密斯-勒米-奥皮茨(SLO)综合征相似。然而,其致死性、多指的常见性以及性器官模糊将这种病症与SLO综合征区分开来。对已发表报告的回顾支持对这种综合征进行单独分类,我们为此提议将其命名为致死性肢端发育不全侏儒症。