The most common clinical presentation of Aeromonas infection is acute gastroenteritis with diarrhea. The extra-intestinal infections caused by aeromonads have been reported as bacteremia, necrotizing fasciitis, peritonitis, meningitis, pneumonia, and arthritis. Herein, we report the first case of acute appendicitis associated with A. sobria and E. coli, which was successfully treated with surgery and antibiotics.