We assessed the retinal microvascular alterations detected by optical coherence tomography angiography (OCT-A) in pediatric and juvenile craniopharyngioma (CP) patients with chiasmal compression. We included 15 eyes of 15 pediatric or juvenile CP patients and 18 eyes of 18 healthy subjects. The evaluation of vessel density from the superficial retinal capillary plexus (SRCP), the deep retinal capillary plexus, and the radial peripapillary capillary (RPC) segments was obtained by OCT-A. The association between vessel density measures and functional and structural measurements was also analyzed. There were significant reductions in the nasal sector of the SRCP (p < 0.0001) and all sectors of the RPC segment vessel density (nasal, temporal, and superior; p < 0.0001, inferior; p = 0.0015) in CP patients postoperatively compared to the healthy subjects. The peripapillary retinal nerve fiber layer (r = 0.6602, p = 0.0074) and ganglion cell-inner plexiform layer thicknesses (r = 0.7532, p = 0.0030) were associated with RPC segment vessel density. Visual acuity (r = − 0.5517, p = 0.0330) and temporal visual field sensitivity loss (r = 0.5394, p = 0.0465) showed an association with SRCP vessel density. In pediatric and juvenile patients with CP, parafoveal and peripapillary vascular changes following chiasmal compression were observed. The changes in vascular structures were closely related to structural and functional outcomes.