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Fig 1.

Kinase binding to type-II kinase inhibitors.

(A) The conformational state of protein kinases (e.g., KDR) including DFG-in (red) and DFG-out (blue) is determined by the DFG-motif. The DFG-pocket (cyan mesh) is unique to the DFG-out conformation. Sorafenib is shown in pink. Broken yellow lines indicate hydrogen bonds. (B) A scheme depicting the positive and negative effects of drug acting on genetic modifiers of medullary thyroid cancer in a Drosophila model. ptc-driven dRetM955T induces lethality during development. ‘Suppressors’ or ‘enhancers’ suppress or enhance, respectively, dRetM955T-induced disease phenotypes as revealed in genetic screening. A drug can suppress lethality by inhibiting the suppressors. It can also induce toxicity and/or worsen transformed phenotypes by inhibiting the enhancers, which results in enhanced lethality.

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Fig 2.

Fly genetics and computational chemistry discovery platform.

Key steps include (A) determining suppressors and enhancers in a dominant modifier genetic screening and their in silico modelability, (B) generating DFG-out kinase models using DFGmodel, (C) virtual screenings of compound libraries against the modeled suppressors and enhancers, combining top-ranking screening results into consensus result, (D) testing top-ranking compounds for rescue of lethality (left panel) and migration of transformed cells in developing wing discs of ptc>dRetM955T flies (right panel), and (E) refining hits by combining structural elements of computationally derived hits and that of drugs and evaluating new targets.

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Fig 3.

Visualization of DFG-pockets.

(A) Electrostatic potential (red, negative potential; blue, positive potential) on the surface of the DFG-pocket in various kinases, including the suppressors RET and SRC, the enhancer TTK, and ERK. (B) Accessible volume of the DFG-pocket (colored volume) for potential type-II kinase inhibitor. Hit molecule 1 is depicted in pink sticks. Broken yellow lines indicate hydrogen bonds.

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Fig 4.

Compound 1 and its analogs.

(A) Rescue of ptc>dRetM955T fly lethality by 1 and 1–1. Both showed improved efficacy (synergy) when co-administrated with 200 μM sorafenib (soraf). (-), vehicle DMSO control. Error bars represent standard error in triplicate experiments. *P < 0.05 in one-sided Student’s t-test as compared with vehicle control. (B) Docking pose of 1 and its analogs 1–1 and 1–2 (salmon sticks) with a DFG-out model of RET (broken yellow lines indicate hydrogen bonds), and their inhibition of migration of the dRetM955T-expressing cells. Right, suppression of cell migration by 1 and 1–1. Controls are shown in (C). (C) In vivo cell migration assay in ptc>dRetM955T flies. Left, a developing whole wing disc containing GFP-labeled, dRetM955T-expressing cells constituting a stripe in the midline. The disc margin is visualized with DAPI (red pseudocolor). There are wild-type cells in black areas. Center, overgrowth of dRetM955T-expressing cells resulting in the thickening of the stripe in the apical view (top). Virtual z-series view of confocal images derived from the plane indicated by yellow dotted lines (bottom) shows dRetM955T-expressing cells migrating away from the original domain (arrows). Right, sorafenib suppressed the migration. White scale bars, 50 μm.

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Fig 5.

Rescue of ptc>dRetM955T flies by 2 and its analogs.

(A) ptc>dRetM955T viability assay. 2 showed increased efficacy when co-administrated with 200 μM sorafenib. (-), vehicle control. Error bars represent standard error in triplicate experiments. *P < 0.05 in one-sided Student’s t-test as compared with no-drug control. (B) Chemical structure of 2 and its analogs. (C) Docking pose of 2 and its analogs in a RET DFG-out model. These compounds are proposed to be putative type-II kinase inhibitors that bind in the DFG-pocket through their 1H-indole moiety and interact with the conserved αC-helix glutamate side chain and DFG-Aspartate backbone (broken yellow lines).

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Table 1.

Kinase inhibition profile of compound 1 at 50 μM.

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Table 2.

Kinase inhibition profile of compounds 2 and 2–3 at 50 μM.

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Fig 6.

Hybrid compounds with improved efficacy.

(A) The kinase inhibitors imatinib, sorafenib, and 2–3 share the common N-phenylcarboxamide moiety (grey box), while the 1H-indole-2-carboxamide of 2–3 resembles the urea linker of sorafenib (blue box). (B) Hybridization of 2 and sorafenib and AD-80 yielded 3 and 4, respectively. Top, chemical structures of compounds. Bottom, docking poses of compounds in a RET DFG-out model. (C) 3 rescued ptc>dRetM955T flies more effectively than by either 2 or sorafenib alone. (-), vehicle control. Error bars represent standard error in triplicate experiments. *P < 0.05 in one-sided Student’s t-test as compared with no-drug control. (D) 3 suppresses migration of dRetM955T-expressing wing disc cells from the original domain (green) similarly to the positive control, sorafenib. Top and bottom, apical and z-series views at the yellow dotted lines in apical view, respectively. Arrows, migrating cells. White scale bars, 50 μm.

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Table 3.

Kinase inhibition profile of compound 3 at 50 μM.

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