PLK4 as a Key Regulator of Neuroblastoma Differentiation and a Promising Therapeutic Target

Int J Biol Sci. 2025 Jul 28;21(11):4979-4996. doi: 10.7150/ijbs.111449. eCollection 2025.

Abstract

Background: Neuroblastoma (NB) differentiation status critically influences prognosis and treatment response. Although differentiation therapy has shown clinical benefit, its efficacy remains limited. The molecular mechanisms driving NB differentiation are not fully understood. PLK4 has been linked to NB tumorigenesis, but its role in regulating differentiation remains unclear. Methods: We investigated the role of PLK4 in neuroblastoma differentiation by modulating its expression both in vitro and in vivo. Through comprehensive analyses employing Western blotting, co-immunoprecipitation, immunofluorescence and murine neuroblastoma models, we identified downstream signaling pathways involved in PLK4-mediated regulation of neuronal genes. Pharmacological inhibition of PLK4 further confirmed its functional relevance in promoting neuroblastoma differentiation. Results: PLK4 functions as a key regulator of neuroblastoma differentiation. Its depletion enhances neuronal maturation and sensitizes cells to 13-cis RA. Mechanistically, we identify a novel PLK4-CXCR4 signaling axis that governs neuroblastoma differentiation through PI3K/Akt-mediated modulation of cyclin D1 expression. The selective PLK4 inhibitor CFI-400945 exhibits dual anti-tumor activity by promoting terminal differentiation and suppressing proliferation. Conclusions: Our study identifies PLK4 as a potential molecular switch governing NB differentiation and a promising therapeutic target to overcome resistance to 13-cis RA.

Keywords: CXCR4; PLK4; cyclin D1; differentiation therapy; neuroblastoma.

MeSH terms

  • Animals
  • Cell Differentiation / drug effects
  • Cell Line, Tumor
  • Child, Preschool
  • Drug Resistance, Neoplasm*
  • Female
  • HEK293 Cells
  • Humans
  • Indazoles
  • Indoles
  • Infant
  • Isotretinoin* / pharmacology
  • Isotretinoin* / therapeutic use
  • Mice
  • Mice, Inbred BALB C
  • Mice, Nude
  • Molecular Targeted Therapy
  • Neuroblastoma* / enzymology
  • Neuroblastoma* / etiology
  • Phosphatidylinositol 3-Kinases / metabolism
  • Protein Serine-Threonine Kinases* / antagonists & inhibitors
  • Protein Serine-Threonine Kinases* / metabolism
  • Proto-Oncogene Proteins c-akt / metabolism
  • Receptors, CXCR4* / metabolism
  • Xenograft Model Antitumor Assays

Substances

  • PLK4 protein, human
  • Protein Serine-Threonine Kinases
  • Isotretinoin
  • 2-(3-(4-((2,6-dimethylmorpholino)methyl)styryl)-1H-indazol-6-yl)-5'-methoxyspiro(cyclopropane-1,3'-indolin)-2'-one
  • Phosphatidylinositol 3-Kinases
  • Proto-Oncogene Proteins c-akt
  • CXCR4 protein, human
  • Receptors, CXCR4
  • Indazoles
  • Indoles