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Developmental biology

Casting an eye over cyclopia

Nature volume 395, pages 112–113 (1998)Cite this article

In ancient Greek mythology the cyclops had a single, central eye. Today, cyclopia is an extreme characteristic of a class of human birth-defect syndromes known as holoprosencephaly. Although the morphological abnormalities that underlie holop rosencephaly are well characterized — a lack of, or defect in, development of the ventral brain — the biochemical defects that underlie this malformation have remained elusive. But the analysis of cyclopic zebrafish (Danio rerio) mutants by Feldman et al.1 and Sampath et al.2 (on pages 181 and 185 of this issue), and by Rebagliati et al.3 in Proceedings of the National Academy of Sciences, provide an insight into the mechanisms that establish the ventral regions of the brain and spinal cord.

Cells in the mesoderm layer at the midline of the embryo secrete signals that establish midline identity in the overlying neural plate. Among these signals are members of the hedgehog (Hh) family of proteins, which can induce a variety of cells, including the neural midline. This region gives rise to the ventral brain of the embryo, and the floorplate and motor neurons in the spinal cord4. Mouse embryos with mutations in the gene that encodes Sonic hedgehog (Shh) do not develop midline structures of the neural plate, leading, at worst, to profound cyclopia. A single, medial eye is located above a nose containing a single nostril5. But although Shh is necessary for specification of the neural plate, other results indicate that it may not be enough. Cloning of the genes responsible for two cyclopic zebrafish mutant phenotypes now suggests that members of the transforming growth factor-β (TGF-β) family may also be involved in these processes.

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Figure 1: Structures formed during embryogenesis.
Figure 2: Alternative models of how nodal and nodal-related factors (Ndr) may act in specification of the floorplate.

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Authors and Affiliations

  1. Institut de Génétique et de la Biologie Moléculaire et Cellulaire, CNRS/INSERM/ULP, BP163, Illkirch Cedex, 67404, CU de Strasbourg, France

    Patrick Blader & Uwe Strähle

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  1. Patrick Blader
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  2. Uwe Strähle
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Blader, P., Strähle, U. Casting an eye over cyclopia. Nature 395, 112–113 (1998). https://doi.org/10.1038/25836

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