BACKGROUND: The zebrafish genetic mutant iguana (igu) has defects in the ciliary basal body protein Dzip1, causing improper cilia formation. Dzip1 also interacts with the downstream transcriptional activators of Hedgehog (Hh), the Gli proteins, and Hh signaling is disrupted in igu mutants. Hh governs a wide range of developmental processes, including stabilizing developing blood vessels to prevent hemorrhage. Using igu mutant embryos and embryos treated with the Hh pathway antagonist cyclopamine, we conducted a microarray to determine genes involved in Hh signaling mediating vascular stability. RESULTS: We identified 40 genes with significantly altered expression in both igu mutants and cyclopamine-treated embryos. For a subset of these, we used in situ hybridization to determine localization during embryonic development and confirm the expression changes seen on the array. CONCLUSIONS: Through comparing gene expression changes in a genetic model of vascular instability with a chemical inhibition of Hh signaling, we identified a set of 40 differentially expressed genes with potential roles in vascular stabilization.
BACKGROUND: The zebrafish genetic mutant iguana (igu) has defects in the ciliary basal body protein Dzip1, causing improper cilia formation. Dzip1 also interacts with the downstream transcriptional activators of Hedgehog (Hh), the Gli proteins, and Hh signaling is disrupted in igu mutants. Hh governs a wide range of developmental processes, including stabilizing developing blood vessels to prevent hemorrhage. Using igu mutant embryos and embryos treated with the Hh pathway antagonist cyclopamine, we conducted a microarray to determine genes involved in Hh signaling mediating vascular stability. RESULTS: We identified 40 genes with significantly altered expression in both igu mutants and cyclopamine-treated embryos. For a subset of these, we used in situ hybridization to determine localization during embryonic development and confirm the expression changes seen on the array. CONCLUSIONS: Through comparing gene expression changes in a genetic model of vascular instability with a chemical inhibition of Hh signaling, we identified a set of 40 differentially expressed genes with potential roles in vascular stabilization.
Authors: Katelynn A Toomer; Mengyao Yu; Diana Fulmer; Lilong Guo; Kelsey S Moore; Reece Moore; Ka'la D Drayton; Janiece Glover; Neal Peterson; Sandra Ramos-Ortiz; Alex Drohan; Breiona J Catching; Rebecca Stairley; Andy Wessels; Joshua H Lipschutz; Francesca N Delling; Xavier Jeunemaitre; Christian Dina; Ryan L Collins; Harrison Brand; Michael E Talkowski; Federica Del Monte; Rupak Mukherjee; Alexander Awgulewitsch; Simon Body; Gary Hardiman; E Starr Hazard; Willian A da Silveira; Baolin Wang; Maire Leyne; Ronen Durst; Roger R Markwald; Solena Le Scouarnec; Albert Hagege; Thierry Le Tourneau; Peter Kohl; Eva A Rog-Zielinska; Patrick T Ellinor; Robert A Levine; David J Milan; Jean-Jacques Schott; Nabila Bouatia-Naji; Susan A Slaugenhaupt; Russell A Norris Journal: Sci Transl Med Date: 2019-05-22 Impact factor: 17.956
Authors: Ankan Gupta; Karthikeyan Thirugnanam; Madhan Thamilarasan; Ashraf M Mohieldin; Hadeel T Zedan; Shubhangi Prabhudesai; Meghan R Griffin; Andrew D Spearman; Amy Pan; Sean P Palecek; Huseyin C Yalcin; Surya M Nauli; Kevin R Rarick; Rahima Zennadi; Ramani Ramchandran Journal: JCI Insight Date: 2022-03-22