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Fig 1.

Structure of CSF1R inhibitor from Janssen.

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Fig 1 Expand

Fig 2.

Upregulation of CSF1R gene expression in IBD patients.

Data is sized by P-value. The data was extracted from Omicsoft® ImmunoLand® 2015 Q4 release [19].

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Fig 2 Expand

Fig 3.

Model of compound 1 with CSF1R.

Modeled using the Schrodinger Maestro Glide docking into a grid generated from the CSF1R X-ray crystal structure (PDB Code: 2I0Y) followed by optimization with MM-GBSA [39].

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Fig 3 Expand

Fig 4.

Prodrug candidates.

a Mean from n ≥3 experiments. b The kinome selectivity data for a panel of 80 kinases are colored by fold selectivity over CSF1R IC50, with ≤10-fold selective in red and a color gradient to ≥100-fold selective in green. c Mean of 1 (compound 1, 4 and 5) or 3 (compound 6) experiments as described in Materials & Methods. d Mean values determined after a 1 mg/kg IV dose (N = 2–3). Data presented as total clearance and unbound clearance (CLb/fub), respectively.

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Fig 4 Expand

Fig 5.

Unbound concentration (ng/mL for plasma; ng/g for colon and liver) of 1 over time after 30 mg/kg PO or PR dosing.

The cellular EC50 corrected for binding in 10% FBS (22 ng/mL) is represented by the horizontal dotted blue line. Tmax = plasma Tmax.

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Fig 5 Expand

Fig 6.

Unbound concentration (ng/mL for plasma; ng/g for colon) of 1 at 3 and 11 h after PO dosing of a 30 mg/kg dose equivalent of prodrugs 2 and 7–14.

Compounds 2, 12, and 13 were dosed as mixtures of isomers (ratio of a:b = 82:18 for 2; 43:57 for 12; and 70:30 for 13). Cmpd 2 is reported as the mean of two separate studies. *Dose = 35 mg/kg, ^Dose = 20 mg/kg. Cellular EC50 corrected for binding in 10% FBS (22 ng/mL) is represented by the horizontal blue line. Tmax = plasma Tmax.

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Fig 6 Expand

Fig 7.

Proposed mechanism of cyclodextrin prodrug cleavage.

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Fig 7 Expand

Fig 8.

Cyclodextrin prodrugs 2 and 12–23.

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Fig 8 Expand

Fig 9.

Unbound concentration (ng/mL for plasma; ng/g for colon) of active parent compounds 1 and 4–6 at 3 and 11 h after PO dosing of a 30 mg/kg dose equivalent of cyclodextrin prodrugs 2, 12, 13, and 16–23.

Prodrugs were dosed to CD1 male (black) or C57BL/6 female (purple) mice (N = 3). Tmax = plasma Tmax. Cellular EC50s for 1, 4, 5, and 6 adjusted for binding in 10% FBS (22, 19, 21, and 42 ng/mL, respectively) are represented by dashed blue lines. *Dose = 25 mg/kg; ^Dose = 32 mg/kg.

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Fig 9 Expand

Fig 10.

Mouse DSS experimental design.

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Fig 10 Expand

Fig 11.

Unbound colon, liver, and blood concentrations of compound 1 in healthy and DSS mice at 11 h (Day 14 DSS) or 12 h (Day 1, Day 7 Healthy) after multi-day dosing of 2.

Also shown are unbound concentrations of compound 3 in DSS mice. Cellular EC50 corrected for binding in 10% FBS is represented by a dashed blue line (compound 1) or a purple line (compound 3).

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Fig 11 Expand

Fig 12.

Depletion of macrophages in the colon and liver by image analysis.

* p<0.05, *** p<0.001, ****p<0.0001; Kruskal-Wallis ANOVA with Dunn’s multiple comparison post-test vs. Vehicle. A subset of animals did not receive the final dose of 2 and were removed from the analysis.

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Fig 12 Expand

Fig 13.

Change in colon erosion length by histology.

*** p<0.001; One-way ANOVA with Dunnett’s multiple comparison post-test vs. Vehicle.

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Fig 13 Expand

Fig 14.

Depletion of circulating monocytes by FACS.

** p<0.01, *** p<0.001, **** p<0.0001; Kruskal-Wallis ANOVA with Dunn’s multiple comparison post-test vs. Vehicle.

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Fig 14 Expand

Fig 15.

Synthesis of primary alcohol 4.

Reagents and conditions: (a) 4-aminopyrazole hydrochloride, Et3N, DMF, 87%; (b) cyclohex-1-en-1-ylboronic acid, 2-(cyclohex-1-en-1-yl)-4,4,5,5-tetramethyl-1,3,2-dioxaborolane, PdCl2dppf, K3PO4, dioxane/water, 90°C, 46%; (c) ethyl 2-bromoacetate, K2CO3, DMF, 90°C, 45% (+ 31% recovered 25); (d) 10% Pd/C, H2 (1 atm), MeOH/EtOAc, rt, 84%; (e) LiAlH4, THF, 0°C to rt, 75%.

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Fig 15 Expand

Fig 16.

Synthesis of secondary alcohol 1.

Reagents and conditions: (a) 4-nitropyrazole, Cs2CO3, DMF, 77% (b) PPTS, acetone/H2O, 88%; (c) NaBH4, EtOH, -5°C; recrystallization from toluene, 59%; (d) Pd/C, H2 (1 atm), MeOH, rt, 98%; (e) 24, DIPEA, DMF, rt, 97%; (f) cyclohex-1-en-1-ylboronic acid, Pd(PPh3)2Cl2, Cs2CO3, 1,4-dioxane/water, 90°C, quant; (g) Pd(OH)2, H2 (1 atm), THF/MeOH/AcOH, rt, 85%.

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Fig 16 Expand

Fig 17.

Synthesis of tertiary alcohol 5.

Reagents and conditions: (a) but-3-en-2-one, Cs2CO3, DMF, rt, 73%; (b) MeMgBr, THF, rt, 38% (+ 44% recovered 33); (c) Pd(OH)2, H2 (1 atm), MeOH/EtOAc/AcOH, rt, 62%.

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Fig 17 Expand

Fig 18.

Synthesis of carboxylic acid 6.

Reagents and conditions: (a) TsCl, pyridine, 0°C to rt, 80%; (b) 4-nitropyrazole, Cs2CO3, DMF, 77%; (c) 10% Pd/C, H2 (1 atm), THF/EtOH, rt, 93%; (d) 24, Et3N, DMF, rt, 97%; (e) cyclohex-1-en-1-ylboronic acid, Pd(PPh3)2Cl2, Cs2CO3, 1,4-dioxane/water, 90°C, 89%; (f) Pd(OH)2, H2 (1 atm), MeOH/THF, rt, 87%; (g) NaOH, MeOH/H2O, rt, 96%.

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Fig 18 Expand

Fig 19.

Synthesis of ester, glucoside and glucuronide prodrugs.

Reagents and conditions: (a) 2-(dimethylamino)acetic acid, EDC, DMAP, DCM, rt, 92%; (b) Boc-(D)-valine, EDC, DMAP, DCM, rt, 72%; (c) TFA, DCM, 0°C to rt, 73% (d) 2,3,4,6-tetra-O-benzyl-alpha-D-glucopyranosyl trichloroacetimidate, BF3.OEt2, -78°C to rt, DCM, 95%; (e) Pd/C, H2 (10 bar), rt, 30% 9, 17% 10; (f) (2R,3R,4S,5S,6S)-2-bromo-6-(methoxycarbonyl)tetrahydro-2H-pyran-3,4,5-triyl triacetate, AgOTf, DCM, 5°C to rt, 7%; (g) LiOH, THF/H2O, 0°C, 50%.

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Fig 19 Expand

Fig 20.

Synthesis of carboxylate-linked cyclodextrin prodrugs.

Reagents and conditions: (a) NaOH, MeCN/H2O, quant; (b) Mono-6-O-(p-toluenesulfonyl)-α-cyclodextrin, DMA, 100°C, 33%; (c) Mono-6-O-(p-toluenesulfonyl)-β-cyclodextrin, DMA, 100°C, 23%; (d) Mono-6-O-mesitylenesulfonyl-γ-cyclodextrin, DMA, 100°C, 27%.

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Fig 20 Expand

Fig 21.

Synthesis of alcohol-linked cyclodextrin and dextran prodrugs.

Reagents and conditions: (a) Et3N, succinic anhydride, DMAP, DCM, rt, 98%; (b) α-cyclodextrin, EDC, DMAP, rt, 32%, 82:18 mix of 2a:2b; (c) β-cyclodextrin, EDC, DMAP, rt, 11%, 42:58 mix of 12a:12b; (d) γ-cyclodextrin, EDC, DMAP, rt, 21% 70:30 mix of 13a:13b; (e) CDI, Et3N, dextran (MW ~70,000) from Leuconostoc spp, DMSO, rt, 27%.

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Fig 21 Expand

Fig 22.

Chemical Synthesis of primary alcohol-linked cyclodextrin prodrugs.

Reagents and conditions: (a) Et3N, succinic anhydride, DMAP, DCM, rt, 71%; (b) α-cyclodextrin, EDCI, DMAP, rt, 22%, 68:32 mix of 16a:16b; (c) β-cyclodextrin, EDCI, DMAP, rt, 5.2% 17a only; (d) γ-cyclodextrin, EDCI, DMAP, rt, 14% 18, 17% 19.

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Fig 22 Expand

Fig 23.

Chemical synthesis of tertiary alcohol-linked cyclodextrin prodrug.

Reagents and conditions: (a) Et3N, succinic anhydride, DMAP, N-hydroxysuccinimide, toluene, 120°C, 54%; (b) α-cyclodextrin, EDCI, DMAP, rt, 22%.

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Fig 23 Expand