Abstract BACKGROUND Craniopharyngioma is histologically benign. However, due to its location, morbidity is high and may be impacted by therapeutic approach. METHODS A retrospective study of patients treated for craniopharyngioma at Seattle Children’s Hospital from 1996-2023 was performed. Surgical approach, extent of resection, subsequent surgery, radiation and medical therapies, and serial surveillance magnetic resonance imaging for progression were reviewed. Five-year overall survival (OS) and progression free survival (PFS) were calculated and morbidity including endocrine and vision dysfunction was described. RESULTS 90 patients were included of which 53(58.9%) were male. Median age at diagnosis was 8.70 (range 1.39-25.17) years. Surgical approach included endoscopic (N=24), craniotomy (N=64), and combined (N=2). Extent of resection was biopsy N=2, subtotal resection N=7, near total resection N=38, gross total resection N=30. Pathology was adamantinomatous subtype in all cases and 68 had a documented CTNNB1 alteration and 3 had a BRAF alteration in those with molecular analysis. 5-year OS was 96.1% (SE 0.13) and 5-year PFS was 51.9% (SE0.21), with median follow-up of 97 months. Transcranial compared to endoscopic endonasal approach was associated with increased risk of progression (62% vs 30%, OR 1.23). 34 patients were treated with radiation upfront (N=19) or at progression (N=15). 55% of patients at diagnosis and an additional 30% in follow up had pituitary hormone dysfunction. Hypothalamic obesity was documented in 31% of patients and varied by surgical approach. At presentation, ophthalmologic abnormalities included optic nerve edema (18%) and optic nerve pallor (24%) and 30% had abnormal visual acuity. Following initial surgery, 50% had improvement in visual acuity, 35% stable and 15% had worsened visual acuity. CONCLUSIONS Upfront surgical strategy may impact risk of disease recurrence; however, its relationship to long term morbidity is not clear. Further work to delineate therapeutic strategies and their relationship to morbidity in this patient population is needed.