Poster Presentation Inaugural Australian Ubiquitin Summit 2025

Ubiquitin Protease Drug Discovery Boosted By Hydrogen Deuterium eXchange Mass Spectrometry (#144)

Benedikt M Kessler 1 , Andrea Pierangelini 1 , Hannah BL Jones 1 , Adán Pinto-Fernández 1 , Andrew J Turnbull 2 , Darragh O'Brien 1
  1. University of Oxford, Headington, Oxford, OXFORDSHIRE, United Kingdom
  2. Cancer Research Horizon, Crick Institute, London, (None), United Kingdom

Recent inclusion of selective DUB inhibitors in clinical trials for the treatment of cancer, kidney and neurodegenerative diseases has heightened interest in DUB-focused drug discovery. We have established a Hydrogen-Deuterium eXchange Mass Spectrometry platform as a tool to capture protein intrinsic disorder in solution [1]. We adapted this approach to profile a panel of DUB – small molecule inhibitor interactions.

We explored the mode of action of covalent USP30 inhibition by a small-molecule containing a cyanopyrrolidine reactive group, USP30-I-1 [2]. The inhibitor demonstrated high potency and selectivity for endogenous USP30 in neuroblastoma cells. Enzyme kinetics and hydrogen-deuterium eXchange mass spectrometry indicated that the inhibitor binds tightly to regions surrounding the USP30 catalytic cysteine and positions itself to form a binding pocket along the thumb and palm domains of the protein, thereby interfering its interaction with ubiquitin substrates. A comparison to a noncovalent USP30 inhibitor containing a benzosulfonamide scaffold [3] revealed a slightly different binding mode closer to the active site Cys77, which may provide the molecular basis for improved potency toward USP30.

We have also applied HDX-MS to determine expanded the repertoire of electrophilic groups targeting DUBs, leading to the discovery of potent and selective inhibitors for USP47, OTUD7B, and USP5 [4]. Using HDX-MS, we reveal the molecular basis of the USP47 inhibitor’s preference over USP7 via selective perturbation of the activation peptide.

Together, we highlight how HDX-MS has been successfully integrated into therapeutic development workflows that can accelerate the development of selective small molecular drug candidates.

  1. [1] Pierangelini A, Kessler BM, O'Brien DP. Driving Therapeutic Innovation in Neurodegenerative Disease With Hydrogen Deuterium eXchange Mass Spectrometry. Mol Cell Proteomics. 2025 Jun 20;24(8):101017. doi: 10.1016/j.mcpro.2025.101017.
  2. [2] O'Brien DP, Jones HBL, Shi Y, Guenther F, Vendrell I, Viner R, Brennan PE, Mead E, Zarganes-Tzitzikas T, Davis JB, Pinto-Fernández A, England KS, Murphy EJ, Turnbull AP, Kessler BM. Structural Dynamics of the Ubiquitin Specific Protease USP30 in Complex with a Cyanopyrrolidine-Containing Covalent Inhibitor. J Proteome Res. 2025 Feb 7;24(2):479-490. doi: 10.1021/acs.jproteome.4c00618
  3. [3] O'Brien DP, Jones HBL, Guenther F, Murphy EJ, England KS, Vendrell I, Anderson M, Brennan PE, Davis JB, Pinto-Fernández A, Turnbull AP, Kessler BM. Structural Premise of Selective Deubiquitinase USP30 Inhibition by Small-Molecule Benzosulfonamides. Mol Cell Proteomics. 2023 Aug;22(8):100609. doi: 10.1016/j.mcpro.2023.100609.
  4. [4] Jones HBL et al., obABPP-HT*: A Precision-Engineered Activity Proteomics Pipeline for the Streamlined Discovery of Deubiquitinase Inhibitors. bioRxiv 2025; doi: https://doi.org/10.1101/2025.05.27.656269