Septic arthritis of the shoulder is very rarely caused by Aeromonas hydrophila. We present the case of a 45-year-old woman who presented with symptoms of painful shoulder for 2 weeks and dropped head for 1 week prior to admission. A. hydrophila was isolated from a culture of purulent synovial fluid. Magnetic resonance imaging revealed profuse abscess collection between the right infraspinatus muscle and trapezius muscle and swelling of the right and left paraspinatus muscles, which suggested myositis-related dropped head syndrome. After surgery with arthrotomy, function of the shoulder and neck extensors was significantly improved.