Mr. Rebagliati et al., CYCLOPS ENCODES A NODAL-RELATED FACTOR INVOLVED IN MIDLINE SIGNALING, Proceedings of the National Academy of Sciences of the United Statesof America, 95(17), 1998, pp. 9932-9937
Ventral structures in the central nervous system are patterned by sign
als emanating from the underlying mesoderm as well as originating with
in the neuroectoderm. Mutations in the zebrafish, Danio rerio, are pro
ving instrumental in dissecting these midline signals. The cyclops mut
ation leads to a loss of medial floor plate and to severe deficits in
ventral forebrain development, leading to cyclopia. Here, we report th
at the cyclops locus encodes the nodal-related protein Ndr2, a member
of the transforming growth factor type beta superfamily of factors. Th
e evidence includes identification of a missense mutation in the initi
ation codon and rescue of the cyclops phenotype by expression of ndr2
RNA, here renamed ''cyclops.'' Thus, in interaction with other molecul
es implicated in these processes such as sonic hedgehog and one-eyed-p
inhead, cyclops is required for ventral midline patterning of the embr
yonic central nervous system.