Aquaporins are selective water channel proteins that play a central role in the homeostasis of human body water. The choroid plexus (CP) is considered to be the main cerebrospinal fluid (CSF)-producing structure. In this study, six specimens of normal human CP obtained during surgery were analyzed by immunohistochemistry techniques for aquaporin-1 (AQP1) expression and distribution. Intense, uniformly distributed AQP1 immunostaining was observable in the apical but not the basolateral side of cuboid cells of the CP. Moreover, this polarized expression of AQP1 was weakly detectable in the endothelial cells of choroid microvessels and, with a different pattern, in the cells lining the tubules shaped into crypts. Selective AQP1 expression on the surface of the normal human CP might explain the role of CSF production by this complex structure.