Targeting alternative splicing as a new cancer immunotherapy-phosphorylation of serine arginine-rich splicing factor (SRSF1) by SR protein kinase 1 (SRPK1) regulates alternative splicing of PD1 to generate a soluble antagonistic isoform that prevents T cell exhaustion

Cancer Immunol Immunother. 2023 Dec;72(12):4001-4014. doi: 10.1007/s00262-023-03534-z. Epub 2023 Nov 16.

Abstract

Background: Regulation of alternative splicing is a new therapeutic approach in cancer. The programmed cell death receptor 1 (PD-1) is an immunoinhibitory receptor expressed on immune cells that binds to its ligands, PD-L1 and PD-L2 expressed by cancer cells forming a dominant immune checkpoint pathway in the tumour microenvironment. Targeting this pathway using blocking antibodies (nivolumab and pembrolizumab) is the mainstay of anti-cancer immunotherapies, restoring the function of exhausted T cells. PD-1 is alternatively spliced to form isoforms that are either transmembrane signalling receptors (flPD1) that mediate T cell death by binding to the ligand, PD-L1 or an alternatively spliced, soluble, variant that lacks the transmembrane domain.

Methods: We used PCR and western blotting on primary peripheral blood mononuclear cells (PBMCs) and Jurkat T cells, IL-2 ELISA, flow cytometry, co-culture of melanoma and cholangiocarcinoma cells, and bioinformatics analysis and molecular cloning to examine the mechanism of splicing of PD1 and its consequence.

Results: The soluble form of PD-1, generated by skipping exon 3 (∆Ex3PD1), was endogenously expressed in PBMCs and T cells and prevents cancer cell-mediated T cell repression. Multiple binding sites of SRSF1 are adjacent to PD-1 exon 3 splicing sites. Overexpression of phosphomimic SRSF1 resulted in preferential expression of flPD1. Inhibition of SRSF1 phosphorylation both by SRPK1 shRNA knockdown and by a selective inhibitor, SPHINX31, resulted in a switch in splicing to ∆Ex3PD1. Cholangiocarcinoma cell-mediated repression of T cell IL-2 expression was reversed by SPHINX31 (equivalent to pembrolizumab).

Conclusions: These results indicate that switching of the splicing decision from flPD1 to ∆Ex3PD1 by targeting SRPK1 could represent a potential novel mechanism of immune checkpoint inhibition in cancer.

Keywords: Immunotherapy; PD1; RNA splicing; SRPK1.

MeSH terms

  • Alternative Splicing*
  • Arginine / genetics
  • Arginine / metabolism
  • B7-H1 Antigen / genetics
  • B7-H1 Antigen / metabolism
  • Cholangiocarcinoma*
  • Humans
  • Immunotherapy
  • Interleukin-2 / genetics
  • Leukocytes, Mononuclear / metabolism
  • Phosphorylation
  • Programmed Cell Death 1 Receptor / metabolism
  • Protein Isoforms / genetics
  • Protein Isoforms / metabolism
  • Protein Serine-Threonine Kinases / genetics
  • RNA Splicing Factors / genetics
  • RNA Splicing Factors / metabolism
  • Serine / chemistry
  • Serine / genetics
  • Serine / metabolism
  • Serine-Arginine Splicing Factors / genetics
  • T-Cell Exhaustion

Substances

  • B7-H1 Antigen
  • Protein Serine-Threonine Kinases
  • RNA Splicing Factors
  • Arginine
  • Serine
  • Interleukin-2
  • Programmed Cell Death 1 Receptor
  • Serine-Arginine Splicing Factors
  • Protein Isoforms
  • SRPK1 protein, human
  • SRSF1 protein, human