Proceedings of the National Academy of Sciences · 1998 · 361 citations · 38 references
Life SciencesDevelopmental BiologyGeneticsDevelopmental NeuroscienceEmbryogenesisMorphogenesisAnterior-posterior Axis FormationDevelopmental Signaling PathwaysDorsal-ventral Axis FormationMorphogen SignalingNeural DevelopmentNeural Tube DevelopmentDevelopmental Patterning In VertebratesEmbryonic Nervous System DevelopmentFloor Plate DevelopmentMidline SignalingMissense MutationsNeural Plate FormationSignalsVentral Patterning
Ventral structures in the central nervous system are patterned by signals emanating from the underlying mesoderm as well as originating within the neuroectoderm. Mutations in the zebrafish, Danio rerio, are proving instrumental in dissecting these midline signals. The cyclops mutation leads to a loss of medial floor plate and to severe deficits in ventral forebrain development, leading to cyclopia. Here, we report that the cyclops locus encodes the nodal-related protein Ndr2, a member of the transforming growth factor type beta superfamily of factors. The evidence includes identification of a missense mutation in the initiation codon and rescue of the cyclops phenotype by expression of ndr2 RNA, here renamed "cyclops." Thus, in interaction with other molecules implicated in these processes such as sonic hedgehog and one-eyed-pinhead, cyclops is required for ventral midline patterning of the embryonic central nervous system.
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Cyclopia and defective axial patterning in mice lacking Sonic hedgehog gene function
Chin Chiang, Ying Litingtung, Eric Lee et al. · Nature · 1996 · 3.1K citations
Mutations in the human Sonic Hedgehog gene cause holoprosencephaly
Erich Roessler, Elena Belloni, Karin Gaudenz et al. · Nature Genetics · 1996 · 1.2K citations