Urban B, Bersu E T
Am J Med Genet. 1987 Jun;27(2):425-34. doi: 10.1002/ajmg.1320270221.
Cases of full and mosaic trisomy 18 and a body of an infant with the 18p-syndrome were dissected in detail to compare the anatomical variations associated with these 3 chromosome imbalances involving autosome 18. The types and numbers of morphologic variations present in both the full and mosaic trisomy 18 bodies were similar to the types and numbers of variations seen in all other cases of full trisomy 18 that have been studied by gross dissection. Apart from an atrial septal defect, the body of the infant with the 18p- imbalance showed only 2 striking defects: 1) deficiencies of the levator palpebrae superioris muscle of the upper eyelid, and 2) absence of the ligament of the head of the femur. The first variation provides a morphologic basis to explain the ptosis which is observed frequently in affected individuals. Absence of the ligament of the head of the femur may be a factor contributing to congenital dislocation of the hip, which is reported occasionally in affected individuals. In addition to providing more detailed information about the phenotype of individual aneuploidy syndromes, studies of cases of different imbalances of single autosomes may provide additional insights about the genotype/phenotype relationships of specific chromosome segments.
对18号染色体完全三体和嵌合三体病例以及一名患有18p综合征婴儿的尸体进行了详细解剖,以比较与这3种涉及18号常染色体的染色体失衡相关的解剖学变异。18号染色体完全三体和嵌合三体尸体中存在的形态学变异类型和数量,与通过大体解剖研究的所有其他18号染色体完全三体病例中观察到的变异类型和数量相似。除房间隔缺损外,患有18p染色体失衡的婴儿尸体仅显示出2个明显缺陷:1)上睑提肌缺陷,2)股骨头韧带缺失。第一个变异提供了一个形态学基础来解释受影响个体中经常观察到的上睑下垂。股骨头韧带缺失可能是导致先天性髋关节脱位的一个因素,在受影响个体中偶尔有报道。除了提供有关单个非整倍体综合征表型的更详细信息外,对单个常染色体不同失衡病例的研究可能会提供有关特定染色体片段基因型/表型关系的更多见解。