OBJECTIVE: To evaluate the prevalence and associated factors involved in cellulitis with lymphangitis among a group of Korean patients who were being treated for lymphedema. We present our epidemiologic research and we also report a systematic review of these types of cases. METHODS: This was a retrospective medical record study among 1,246 patients diagnosed with lymphedema. The study was carried out between January 2006 and December 2012 at the Kosin University Gospel Hospital and Seoul National University Bundang Hospital. Cases were examined for onset time, affected site, seasonal trend, and recurrence pattern of lymphedema, lymphangitis, and cellulitis. We also evaluated the history of blood-cell culture and antibiotic use. RESULTS: Ninety-nine lymphedema patients experienced complications such as cellulitis with accompanying lymphangitis. Forty-nine patients had more than two recurrences of cellulitis with lymphangitis. The incidence and recurrence of cellulitis with lymphangitis were significantly higher in the patients with lower-extremity lymphedema. There was a significant trend toward higher cellulitis prevalence in the lower-extremity lymphedema group according to the time of lymphedema onset. Among the cellulitis with lymphangitis cases, 62 cases were diagnosed through blood-cell culture; 8 of these 62 cultures were positive for β-hemolytic streptococci. CONCLUSION: The prevalence rate of cellulitis with lymphangitis in patients with lymphedema was 7.95%, and the prevalence of recurrent episodes was 3.93%. Especially, there was high risk of cellulitis with lymphangitis after occurrence of lower-extremity lymphedema with passage of time. Lymphedema patients should be fully briefed about the associated risks of cellulitis before treatment, and physicians should be prepared to provide appropriate preventive education.