A 10-year-old girl presented with esotropia. She had 35 prism diopters of esodeviation at far and near with left inferior oblique muscle overaction (+3). Cycloplegic refraction was +0.5D OD and +0.75D OS, and visual acuity was 20/25 in each eye. The patient was being treated with G-CSF (Leufokine^?) due to her severe congenital neutropenia (WBC 3350, neutrophil 7.3%, and ANC 170/ul). Despite of low ANCs, we decided to perform a surgery because the patient and her parents strongly wanted it. Prophylactic topical antibiotics (Cravit^?, levofloxacin 0.5%) were applied four times a day for three days prior to the surgery. We sterilized the periocular skin with povidone-iodine 10% and placed povidone-iodine 5% drops in the cul-de-sac preoperatively. Strabismus surgery was performed. After surgery, profuse irrigation with the entire bottle of topical antibiotics (Vigamox^?, moxifloxacin hydrochloride 0.5%) was performed for each eye. We prescribed topical and highly potent systemic antibiotics for 1 week. This is the first case report to describe the successful perioperative antisepsis care following strabismus surgery for a patient with severe congenital neutropenia. Prevention of postoperative infection in granulocytopenic patients could be achieved using antibiotic agents which provide the greatest spectrum of coverage against both Gram-positive and Gram-negative infections.