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Lipedema is a chronic disease characterized by the disproportionate and symptomatic accumulation of fat in the lower limbs and arms. Women with lipedema experience heaviness, fatigue and pain, leading to impairments in daily activities and reduced quality of life. Conservative treatment mainly focuses on lifestyle modifications, along with compression therapy and manual lymphatic drainage. Exercise training could also play a pivotal role in the management of lipedema. The aim of this systematic review was to explore the effectiveness of exercise training in women with lipedema. Acomprehensive literature search was conducted in Embase, MEDLINE, Cochrane CENTRAL, Web of Science, CINAHL on June 23, 2025. The main outcomes were pain, fatigue and other symptoms, quality of life, anthropometric characteristics, lower limb volumes and/or circumference, and functional performance. The search strategy identified 523 studies, of which six were included in this review. Studies included 115 women with lipedema. Exercise training seems to improve pain and other symptoms, quality of life, limb volumes and/or circumference, and functional performance. The effects seem to be greater when combined with ongoing compression therapy. However, the effects of exercise training on the different outcomes varied among the studies, probably related to the high heterogeneity, different training approaches, and small sample sizes. Exercise training might be a promising therapeutic care option in women with lipedema, and the effects seem to be greater when combined with ongoing compression therapy. Appropriately designed and adequately powered studies are needed to further explore the benefits of exercise in these patients. (PROSPERO Registration No.: CRD42024604164)