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Neutropenia is a rare complication of drug therapy and is usually underdiagnosed. Cefoperazone/sulbactam is a combination of broad-spectrum antibacterial agents. Data on cefoperazone/sulbactam-induced neutropenia are limited. Herein, we report the case of a 35 year-old female patient who was admitted to the hospital due to an appendiceal abscess. After anti-infective treatment with cefoperazone/sulbactam, the patient developed neutropenia on day 4. After discontinuing treatment with cefoperazone/sulbactam, the patient’s white blood cells and neutrophils gradually returned to normal. Hence, clinicians should monitor changes in neutrophil count during cefoperazone/sulbactam therapy and provide timely treatment.