Aim. The aim of the study was to reveal structural peculiarities of the ventricular germinal zone and of the neocortex in newborns of 22-40 weeks gestation with congenital hydrocephalus associated with anomalies in development of the cerebral aqueduct.
 Materials and Methods. The main group was the brain of newborns aged 22-40 weeks of gestation with hydrocephalus, formed with the underlying developmental anomalies of the cerebral aqueduct (n=10); comparison group (n=30) was the brain of newborns without visible pathology of the CNS with the width of the lumen of lateral ventricles not exceeding 0.5 cm. A complex pathomorphological study of the ventricular germinal zone and the neocortex in the projection of field No. 6 (cortical representation of the motor analyzer) was carried out.
 Results. Hydrocephalus associated with anomalies of development of the cerebral aqueduct is characterized by disorders in formation of sulci and gyri (micropolygyria, asymmetry and breakage of the gyrification sequence), by absence of signs of remodeling of the ventricular germinal zone and by retardation of differentiation of III-VI cytoarchitectonic layers of the neocortex, with the underlying increased expression of S-100 glial protein in glioblasts of white matter, decreased expression of vimentin in vessels of periventricular region, negative expression of metalloproteinases-9 in glioblasts, of reelin in Cajal-Retzius neurons, of desmin in periventricular vessels.
 Conclusion. Structural peculiarities of the ventricular germinal zone and the neocortex in newborns of 22-40 weeks of gestation with hydrocephalus, associated with anomalies in development of the cerebral aqueduct, should be considered as morphological diagnostic criteria for differentiation of the given pathology with internal hydrocephalus of a different etiology.