Background: To retrospectively reported a case of bone marrow hyperplasia in elderly acute lymphoblastic leukemia changed from low into hyperactive, in order to improve the understanding of this disease. Case presentation: We continuously followed up a patient of bone marrow hypoplastic patients by evaluating his clinical results, bone marrow morphology and flow cytometry, and analyzing the results at different locations in two consecutive bone marrow punctures. The patient’s hypoplastic bone marrow suddenly became extremely hyperplasia. Before this change, there was early-stage alterations in morphology, flow cytometry and biopsy of the bone marrow, but there was no significant change in peripheral blood cells.Conclusions: For patients whose hypoplastic bone marrow suddenly became extremely hyperplasia, morphology was not very useful, thus definite diagnosis must be dependent on immunological types and bone marrow biopsy results.