We herein report a case of peritoneal dialysis-associated peritonitis caused by Lysinibacillus sphaericus in a 40s-year-old patient. Treatment was initiated with intermittent intraperitoneal cefazolin and ceftazidime. Later, both peritoneal dialysate and blood cultures detected L. sphaericus, so the antibiotic was changed to ampicillin (ABPC). The patient was treated with a combination of intraperitoneal intermittent and intravenous ABPC for 7 days, followed by 14 days of amoxicillin. The patient experienced no adverse events and no recurrence for 30 days. The patient had four dogs, and the infection was deemed likely to have been caused by environmental contamination and inadequate catheter replacement.

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