The renal lineage factor PAX8 controls oncogenic signalling in kidney cancer

Nature. 2022 Jun;606(7916):999-1006. doi: 10.1038/s41586-022-04809-8. Epub 2022 Jun 8.

Abstract

Large-scale human genetic data1-3 have shown that cancer mutations display strong tissue-selectivity, but how this selectivity arises remains unclear. Here, using experimental models, functional genomics and analyses of patient samples, we demonstrate that the lineage transcription factor paired box 8 (PAX8) is required for oncogenic signalling by two common genetic alterations that cause clear cell renal cell carcinoma (ccRCC) in humans: the germline variant rs7948643 at 11q13.3 and somatic inactivation of the von Hippel-Lindau tumour suppressor (VHL)4-6. VHL loss, which is observed in about 90% of ccRCCs, can lead to hypoxia-inducible factor 2α (HIF2A) stabilization6,7. We show that HIF2A is preferentially recruited to PAX8-bound transcriptional enhancers, including a pro-tumorigenic cyclin D1 (CCND1) enhancer that is controlled by PAX8 and HIF2A. The ccRCC-protective allele C at rs7948643 inhibits PAX8 binding at this enhancer and downstream activation of CCND1 expression. Co-option of a PAX8-dependent physiological programme that supports the proliferation of normal renal epithelial cells is also required for MYC expression from the ccRCC metastasis-associated amplicons at 8q21.3-q24.3 (ref. 8). These results demonstrate that transcriptional lineage factors are essential for oncogenic signalling and that they mediate tissue-specific cancer risk associated with somatic and inherited genetic variants.

MeSH terms

  • Alleles
  • Basic Helix-Loop-Helix Transcription Factors / metabolism
  • Carcinogenesis* / genetics
  • Carcinoma, Renal Cell / metabolism
  • Carcinoma, Renal Cell / pathology
  • Cyclin D1 / genetics
  • Gene Expression Regulation, Neoplastic
  • Humans
  • Kidney / metabolism
  • Kidney / pathology
  • Kidney Neoplasms* / metabolism
  • Kidney Neoplasms* / pathology
  • Mutation
  • PAX8 Transcription Factor* / genetics
  • PAX8 Transcription Factor* / metabolism
  • Proto-Oncogene Proteins c-myc / genetics
  • Signal Transduction*
  • Von Hippel-Lindau Tumor Suppressor Protein / genetics

Substances

  • Basic Helix-Loop-Helix Transcription Factors
  • CCND1 protein, human
  • MYC protein, human
  • PAX8 Transcription Factor
  • PAX8 protein, human
  • Proto-Oncogene Proteins c-myc
  • Cyclin D1
  • endothelial PAS domain-containing protein 1
  • Von Hippel-Lindau Tumor Suppressor Protein
  • VHL protein, human