Lymphedema is a distinct form of edema characterized by its chronic and progressive course, resulting from impaired lymphatic formation and drainage. Both congenital and acquired abnormalities of the lymphatic system can lead to the accumulation of macromolecules within the interstitial space and subsequent fluid retention. Lymphorrhea accompanying congenital lymphedema is rare, and studies evaluating its effects on muscle oxygenation are limited in the literature. This case report describes the effects of a comprehensive rehabilitation program in a 52-year-old woman with advanced congenital lymphedema and lymphorrhea, emphasizing changes in muscle oxygenation. The patient, who had lifelong lower limb swelling and mobility restriction, was diagnosed with stage 2 congenital lymphedema and lymphorrhea and participated in a six-week rehabilitation program comprising manual lymphatic drainage, skin care, exercise, and multilayer compression bandaging performed five times per week. Following treatment, limb circumference decreased, joint range of motion improved, and muscle oxygenation assessed by near-infrared spectroscopy showed notable enhancement. Additionally, the patient lost 12 kg, was able to walk 1 km without pain, and reported an improved quality of life. These findings demonstrate that comprehensive rehabilitation approaches involving manual lymphatic drainage can significantly contribute to not only edema control but also improved muscle oxygenation and functional capacity in congenital lymphedema with lymphorrhea.