The incidence of tuberculosis in patients treated with hemodialysis for chronic renal failure is higher than in the general population. Further, those patients often experience adverse effects from therapy with anti-tuberculosis drugs. Although the ototoxicity and toxic retinopathy caused by antituberculosis drugs is well known, the cerebellar syndrome is rare. We presented a 50-year-old woman developed cerebellar ataxia who was on hemodialysis program and taking anti-tuberculosis drugs combination therapy. Brain magnetic resonance imaging showed cerebellar edema. Cerebellar signs partially reversed after discontinuation of anti-tuberculosis therapy and initiation of pyridoxine therapy. In our patient, isoniazid was probably the drug that caused cerebellar signs and symptoms. In conclusion, a diagnosis of isoniazid-induced cerebellitis should be considered when cerebellar signs develop in patients undergoing hemodialysis and treated with isoniazid.