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Title

Recurrent multiple CNS hemangioblastomas with VHL disease treated with pazopanib: a case report and literature review
Authors
Migliorini, Denis
Haller, Sven
Merkler, Doron
Pugliesi-Rinaldi, Angela
Koka, Avinash
Schaller, Karl Lothard
Leemann, Béatrice
Dietrich, Pierre-Yves
Published in CNS Oncology. 2015, vol. 4, no. 6, p. 387-92
Abstract 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.
Identifiers
PMID: 26497655
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Structures
Faculté de médecine / Section de médecine clinique / Département de médecine interne des spécialités
Faculté de médecine / Section de médecine fondamentale / Département de pathologie et immunologie
Faculté de médecine / Section de médecine clinique / Département des neurosciences cliniques
Faculté de médecine / Section de médecine clinique / Département de radiologie et informatique médicale
Research groups La Sclérose en plaques (908)
Immunothérapie des cancers (42)
Citation
(ISO format) 		MIGLIORINI, Denis et al. Recurrent multiple CNS hemangioblastomas with VHL disease treated with pazopanib: a case report and literature review. In: CNS Oncology, 2015, vol. 4, n° 6, p. 387-92. https://archive-ouverte.unige.ch/unige:84085

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Deposited on : 2016-05-31

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