Background. Congenital knee dislocation is a rare disease of the musculoskeletal system (1 in 100,000 live births). In the literature, few studies have described the anatomical changes characteristic of this pathology, which are only revealed during surgical treatment. Aim. This study aimed to evaluate the pathomorphological features of the ligamentous–articular apparatus and thigh muscles with congenital knee dislocation on autopsy material. Materials and methods. The study included two fetuses with bilateral congenital knee dislocation after spontaneous miscarriage at 18 and 20 weeks of gestation and one stillborn fetus with bilateral congenital knee dislocation at 29 weeks of gestation. The comparison group was composed of two fetuses after spontaneous miscarriages at 18 and 20 weeks of gestation and one stillborn fetus at 25 weeks of gestation without anomalies of the lower extremities. Results. Various abnormalities and displacements of the anatomical structures, as well as degenerative dystrophic changes in the soft tissues during histological examination, were found. Pathomorphological changes in the control group were not detected. Conclusion. Pathomorphological changes are the main manifestations of congenital knee dislocation in the studied fetuses.