The long-term objective of this proposal is to gain a detailed understanding of the genetic control of eye-tissue determination in Drosophila. The specification of the eye primordium involves interactions between multiple signaling pathways (dpp, wg, hh) and transcription factors (Toy, Ey, Eya, So, Dae, Hth, Tsh) linked in a regulatory cascade that controls initiation of neurogenesis in the eye. In this grant, we propose to investigate the function of these molecules in initiating neurogenesis, to analyze the role of a novel gene in this process, and to identify other factors that contribute to eye specification. ? ? The specific aims of this grant are: ? 1. to elucidate the molecular mechanisms leading to initiation of neurogenesis by dissecting the transcriptional control of the proneural gene atonal; ? 2. to investigate at molecular and genetic levels the function of l(3)96Bb-CG5807, a locus required for eye specification; ? 3. to identify novel regulators of eye specification relying on a modified misexpression screen. ? ? Since the factors studied in this proposal are conserved and play significant roles in vertebrate eye development, our work will provide insights into genetic factors that are central to mammalian eye development and are linked to congenital disorders of the eye and brain. ? ? ?

National Institute of Health (NIH)
National Eye Institute (NEI)
Research Project (R01)
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Biology and Diseases of the Posterior Eye Study Section (BDPE)
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Mariani, Andrew P
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Massachusetts Eye and Ear Infirmary
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Zhou, Qingxiang; Yu, Linlin; Friedrich, Markus et al. (2017) Distinct regulation of atonal in a visual organ of Drosophila: Organ-specific enhancer and lack of autoregulation in the larval eye. Dev Biol 421:67-76
Zhou, Qingxiang; DeSantis, Dana F; Friedrich, Markus et al. (2016) Shared and distinct mechanisms of atonal regulation in Drosophila ocelli and compound eyes. Dev Biol 418:10-16
Yu, Linlin; Zhou, Qingxiang; Pignoni, Francesca (2015) ato-Gal4 fly lines for gene function analysis: Eya is required in late progenitors for eye morphogenesis. Genesis 53:347-55
Moody, Sally A; Neilson, Karen M; Kenyon, Kristy L et al. (2015) Using Xenopus to discover new genes involved in branchiootorenal spectrum disorders. Comp Biochem Physiol C Toxicol Pharmacol 178:16-24
Zhou, Qingxiang; Zhang, Tianyi; Jemc, Jennifer C et al. (2014) Onset of atonal expression in Drosophila retinal progenitors involves redundant and synergistic contributions of Ey/Pax6 and So binding sites within two distant enhancers. Dev Biol 386:152-64
Pina, Cara; Pignoni, Francesca (2012) Tubby-RFP balancers for developmental analysis: FM7c 2xTb-RFP, CyO 2xTb-RFP, and TM3 2xTb-RFP. Genesis 50:119-23
Yu, Linlin; Zhou, Qingxiang; Zhang, Chuanxi et al. (2012) Identification of Bombyx atonal and functional comparison with the Drosophila atonal proneural factor in the developing fly eye. Genesis 50:393-403
Bras-Pereira, Catarina; Zhang, Tianyi; Pignoni, Francesca et al. (2011) Homeostasis of the Drosophila adult retina by actin-capping protein and the Hippo pathway. Commun Integr Biol 4:612-5
Zhang, Tianyi; Zhou, Qingxiang; Pignoni, Francesca (2011) Yki/YAP, Sd/TEAD and Hth/MEIS control tissue specification in the Drosophila eye disc epithelium. PLoS One 6:e22278
Neilson, Karen M; Pignoni, Francesca; Yan, Bo et al. (2010) Developmental expression patterns of candidate cofactors for vertebrate six family transcription factors. Dev Dyn 239:3446-66

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