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¹Eskişehir Osmangazi Üniversitesi, Tıp Fakültesi, Beyin ve Sinir Cerrahisi Anabilim Dalı, Eskişehir, Türkiye Craniopharyngiomas have long been considered a benign tumor but the treatment is very challenging and oftentimes only associated with poor patient outcome and very high morbidity. Improved treatment strategies can only be achieved with better understanding of the disease biology. Recent molecular biological findings on the molecular biology of craniopharyngiomas have improved our understanding. In this paper, current understanding of craniopharyngioma molecular biology that has made its way to the clinic is reviewed. Different molecular derangements have been discovered in adamantinomatous and papillary craniopharyngiomas. The adamantinomatous type is associated with CTNNB1 gene mutations in 92-96% of the cases whereas the papillary type is associated with BRAF V600E gene mutations in 95-100% of the cases. Both mutations are associated with derangement of different intracellular signaling cascades. Today, craniopharyngioma must be considered as a complex disease with a molecular pathogenesis and novel treatment strategies shall be developed with this notion in mind. Anahtar Kelimeler : Craniopharyngioma, Papillary, Adamantinomatous, Molecular biology