Hemangioblastoma is a rare benign neoplasm, accounting for less than 2% of all 
primitive brain tumors. It may arise sporadically in a solitary form, or 
associated with Von Hippel-Lindau (VHL) disease with multiple tumors. Surgery is 
the mainstay treatment, but management is challenging in case of recurrent 
and/or multiple tumors. VHL protein is defective in both forms of 
hemangioblastoma, leading to the accumulation of hypoxia-inducible factor, 
stimulating angiogenesis via VEGF and PDGF mainly. Here, we report a 37-year-old 
woman's case with recurrent and rapidly progressive VHL-associated 
hemangioblastomas, causing severe disability. She was treated 24 months with 
pazopanib, a multityrosine kinase inhibitor (TKI) targeting VEGF and PDGF-β 
pathways. Despite moderate radiological changes, progressive improvement in her 
clinical condition persisting over 3 years was observed. Inhibiting angiogenesis 
is a therapeutic option that may improve the quality of life and the autonomy of 
VHL patients disabled with multiple hemangioblastomas.