We recently reported the discovery of a recurrent “hotspot” somatic missense mutation of the GPCR gene CYSLTR2. The mutant CYSLTR2 encodes cysteinyl-leukotriene receptor 2 (CysLTR2)–L129Q that carries a single amino acid substitution at a highly conserved residue in helix 3 (Ballesteros–Weinstein generic position 3.43) (3) and serves as a driver oncogene in patients with uveal melanoma (UVM) (2). This evidence concerns the gene CYSLTR2 and uveal melanoma.