The transcription factor Pax6 is considered the master control gene for eye formation because (1) it is present within the genomes and retina/lens of all animals with a visual system; (2) severe retinal defects accompany its loss; (3) Pax6 genes have the ability to substitute for one another across the animal kingdom; and (4) Pax6 genes are capable of inducing ectopic eye/lens in flies and mammals. Many roles of Pax6 were first elucidated in Drosophila through studies of the gene eyeless (ey), which controls both growth of the entire eye-antennal imaginal disc and fate specification of the eye. We show that Ey also plays a surprising role within cells of the peripodial epithelium to control pattern formation. It regulates the expression of decapentaplegic (dpp), which is required for initiation of the morphogenetic furrow in the eye itself. Loss of Ey within the peripodial epithelium leads to the loss of dpp expression within the eye, failure of the furrow to initiate, and abrogation of retinal development. These findings reveal an unexpected mechanism for how Pax6 controls eye development in Drosophila.

Author contributions

Conceptualization: J.P.K.; Formal analysis: L.R.B., B.M.W., S.D.N., J.P.K.; Investigation: L.R.B., B.M.W., A.N., S.D.N.; Writing - original draft: J.P.K.; Writing - review & editing: L.R.B., B.M.W., A.B., J.P.K.; Supervision: J.P.K.; Project administration: J.P.K.; Funding acquisition: A.B., J.P.K.

Funding

This work is supported by grants from the National Institute of General Medical Sciences (R01 GM123204 to A.B.) and from the National Eye Institute (R01 EY014863 to J.P.K.). Deposited in PMC for release after 12 months.

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