Background: Neurosurgeries are at high risk of surgical site infections (SSI), a complication associated with increased morbidity, mortality, and cost. Our aim was to measure SSI incidence and risk factors following pediatric neurosurgery at CHU Sainte-Justine, the provincial center for pediatric craniofacial surgery in Québec, Canada. Methods: Retrospective cohort study of all patients with elective neurosurgery performed at CHUSJ between October 2014 and October 2018. Medical records were reviewed to compare demographics, clinical presentations, and outcomes of patients. SSIs occurring within 30 days of a procedure without implant and up to 90 days with implant, were identified. SSI incidence was measured in patient years, and risk factors were assessed using univariate logistic regressions. Results: In total, 379 patients were included with an overall SSI incidence of 3.96 patient years. We found a higher SSI incidence in 2014–2015 compared to 2016–2018 (1.82 vs 4.83 patient years). The median age was 3.90 years, and cases seemed younger than controls (1.45 vs 4.15 years). No difference between groups was found for sex, body mass index, prematurity, and length of hospitalization. The proportion of deep SSIs was greater than superficial SSIs (53.3% vs 46.7%). Cases were more likely to present with a more severe ASA score, previous history of neurosurgery, neurological conditions, and pulmonary conditions than controls: OR, 3.90 (95% CI, 1.36–11.49); OR, 2.59 (95% CI, 0.88–7.40); OR, 2.77 (95% CI, 0.98–8.41), and OR, 3.21 (95% CI, 0.86–9.94), respectively. Among patients with history of neurosurgery, a higher proportion of cases experienced a cerebrospinal fluid leak (28.6% vs 2.2%). Most patients (85.8%) received preoperative prophylactic antibiotic. Of those, 49.3% were considered appropriate based on antibiotic and timing of administration. When antibiotic dosage was also considered, the number of patients who received an appropriate antibiotic therapy decreased radically. Conclusions: Patients with comorbidities, especially neurological and pulmonary conditions, are at higher risk of SSI after neurosurgery. We are currently working on a detailed analysis to explain the increase in SSI incidence after 2016. Finally, prophylactic antibiotic therapy needs to be improved and its impact on SSI rates needs to be monitored.