Headache is the most common symptom impacting the quality of life of patients with Chiari I malformation. While headaches typically present in the occipital or suboccipital regions, and induced by Valsalva maneuver, complex migraine-like headaches referring to frontal or orbital regions also occur. Our study explores the therapeutic potential of C1 nerve root decompression in addition to increasing intracranial compliance in patients with Chiari I malformation. We retrospectively analyzed cases operated on for Chiari I malformation, focusing on patients with non-suboccipital, non-Valsalva induced headaches. Preoperative and postoperative visual analog scale (VAS) scores and headache frequency were recorded. Surgical technique involved suboccipital craniectomy, C1 laminectomy, cerebellar tonsil reduction, and C1 root decompression. Twelve patients (10 females, 2 males) aged 22-52 were included. Preoperative mean VAS score was 8, which improved to 2 or less in 11 patients postoperatively. The mean headache frequency decreased significantly. Our findings suggest that C1 root compression contributes to atypical headaches in Chiari I malformation and that C1 root decompression may improve surgical outcomes.