Objectives:Early-life epilepsies (epilepsies in children 1 to 36 months old) are common and may be refractory to anti-seizure medications. We summarize findings of a systematic review commissioned by the American Epilepsy Society to assess evidence and identify evidence gaps for surgical treatments for epilepsy in children aged 1 to 36 months without infantile spasms.Methods:EMBASE, MEDLINE, PubMed, and the Cochrane Library were searched for studies published from 1/1/1999 to 8/19/21. We included studies reporting data on children aged 1 month to ≤ 36 months undergoing surgical interventions or neurostimulation for epilepsy and enrolling ≥ 10 patients per procedure. We excluded studies of infants with infantile spasms or status epilepticus. For effectiveness outcomes (seizure freedom, seizure frequency), studies were required to report follow-up at ≥ 12 weeks. For harm outcomes, no minimum follow-up was required. Outcomes for all epilepsy types, regardless of etiology were reported together.Results:Eighteen studies (in 19 articles) met inclusion criteria. Sixteen pre/post studies reported on efficacy and twelve studies addressed harms. Surgeries were performed from 1979 to 2020. Seizure freedom for infants undergoing hemispherectomy/hemispherotomy ranged from 7% to 76% at 1 year after surgery. For non-hemispheric surgeries seizure freedom ranged from 40% to 70%. For efficacy, we concluded low strength of evidence suggests some infants achieve seizure freedom after epilepsy surgery. Over half of infants undergoing hemispherectomy/hemispherotomy achieved a favorable outcome (Engel I or II, ILAE I to IV, or >50% seizure reduction) at follow-up of >1 year, although studies had key limitations.Surgical mortality was rare for functional hemispherectomy/hemispherotomy, and non-hemispheric resections. Low strength of evidence suggests post-operative hydrocephalus is uncommon for infants undergoing non-hemispheric procedures for epilepsy.Conclusion:Although existing evidence remains sparse and low quality, some infants achieve seizure freedom after surgery and ≥50% achieve favorable outcomes. Future prospective studies in this age group are needed. In addition to seizure outcomes, studies should evaluate other important outcomes (developmental outcomes, quality of life [QOL], sleep, functional performance, and caregiver QOL).Registration:This systematic review was registered in PROSPERO (CRD42021220352) on March 5, 2021.