Genital lymphoedema is a rare but debilitating and disfiguring complication of longstanding hidradenitis suppurativa (HS). Despite the existence of medical and surgical methods that offer varying success rates in a limited number of cases, no data exist about the use of complex decongestive therapy (CDT) in HS-related genital lymphoedema. This case report describes the treatment and outcome of a 56-year-old male patient with severe scrotal lymphoedema due to underlying HS (Hurley stage 3). The patient was unresponsive to various topical and systemic antibiotics and biological agents, including adalimumab and certolizumab pegol. When the patient was assessed, ixekizumab treatment for his HS was planned. He had progressive oedema in the genital area for two years with difficulty in wearing trousers and having sexual intercourse, and painful urination. CDT was recommended for three days a week concurrently with ixekizumab treatment. The patient and his wife were also educated about self-drainage techniques and skincare maintenance. After six sessions of CDT over 14 days, the patient demonstrated a significant reduction in scrotal measurements. He achieved a better scrotal contour, the degree of the buried penis was decreased, and urination was easier and painless. The findings of this case report showed that CDT was an easily applicable, practical and promising method that offered a rapid treatment response for HS-related genital lymphoedema.:10.12968.33.Sup2a.xxviii