Erythropoiesis-stimulating agents (ESAs) have been the mainstay of renal anemia treatment. However, an unmet clinical need remains owing to the ESAs hyporesponsiveness. Bone marrow hypoplasia is one of the important mechanisms contributing to ESAs hyporesponsiveness. Herein we report a middle-aged female patient undergoing maintenance hemodialysis with ESAs hyporesponsiveness and bone marrow hypoplasia. Before hospitalization, a high dose of ESA supplement (r-HuEPO 16000 U/week) was given, but the patient seemed unresponsive (haemoglobin level 5.9 g/dL). Then, the patient’s medication was switched from r-HuEPO to roxadustat at a dosage of 120 mg three times per week. Unfortunately, she still showed a poor response to the conventional recommended dose of roxadustat. Given the patient’s past medical history and clinical use of roxadustat, we prescribed a dose of roxadustat of 150 mg three times per week, and the haemoglobin level increased gradually to 9.8 g/dL after 1 month. Taken together, we report the case of an anemic patient with bone marrow hypoplasia who exhibited a poor response to high-dose ESAs. Although the dosage of roxadustat used was higher than the conventional recommended dosage, it was shown that roxadustat could correct renal anemia effectively and safely. Therefore, this case provides unique experience for the use of this new class of drug to treat patients with ESA hyporesponsiveness.