Abstract: Objective To improve the understanding of drug-induced pure red cell aplasia(PRCA). Methods A case of isoniazidinduced PRCA was reported. The features, diagnosis and treatment of isoniazid-induced PRCA were analyzed. The case was a 94 years old man who was diagnosed with pulmonary tuberculosis(TB) and chronic kidney disease(CKD). Progressive anemia occurred during his treatment was finally diagnosed as PRCA by bone marrow biopsy, which was initially misdiagnosed as anti-EPO antibodyinduced PRCA. Results Finally we found that PRCA was induced by isoniazid. Conclusion This case provides the clinical evidence for unexplained anemia occurred during the drug therapy for TB and isoniazid may induce PRCA.