One case of melioidosis associated with rare brain lesion and secondary epilepsy was reported, patient was admitted to a hospital twice because of "right hip pain for 10 days" and "epileptic seizure", Burkholderia pseudomallei was isolated through blood culture,and was diagnosed as melioidosis, antimicrobial susceptibility testing showed that it was susceptible to imipenem and compound sulfamethoxazole, magnetic resonance imaging (MRI) of brain proved that it was multiple brain lesions in right centrum ovale, corpus callosum, and posterior cingulate gyrus. After 6 months of symptomatic medical treatment, including anti-infective treatment and organ function support, patient recovered well. Therefore, relevant examination should be conducted in time for melioidosis complicated with central nervous system diseases, including cranial MRI, active bacterial culture and antimicrobial susceptibility testing, as well as long-term standardized anti-infective treatment, so as to avoid recurrence.