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Chronic exertional compartment syndrome (CECS) presents a unique therapeutic challenge. Fasciotomy, currently the most well accepted treatment approach, still has a significant number of treatment failures, demonstrating the need for additional options. Botulinum toxin has been introduced as a potential therapeutic agent, but long-term outcomes are unknown. We present the longest documented follow-up (14 months) of a CECS case treated with botulinum toxin injections. At 14 months follow-up, the patient reported continued pain relief and had resumed her active lifestyle without any adverse effects. Although more research is needed to optimize patient selection and treatment protocol, this case illustrates the potential for botulinum toxin as a long duration, low risk alternative treatment option for CECS.