This study was purpose to investigate the cytologic features of bone marrow (BM) and peripheral blood (PB) in hyperleukocytic acute leukemia (HAL) and their clinical significance in accordance with high leukocyte count as poor prognostic factor for acute leukemia. The smears of BM and PB were collected from 68 out-patients and inpatients including 28 cases of HLA and 40 cases of non HAL (NHAL) in our hospital since 2009. The proliferation degree, morphology and abnormal appearance in each cell lineage were observed with HE, POX, PAS, NSE+ NaF staining for BM mears and HE staining for PB smears by means of optical microscope. The final diagnosis was made by cellular chemical staining results, then the counting and classification were performed in 200 nucleated cells to calculate the percentage of each cell lineage, the myeloid/erythroid ratio and so on. The BP smears were observed with the same methods, the counting and classification of 100 nucleated cells were performed to calculate a variety of nucleated cell percentage. The resulted data were analyzed by the SPSS 12.0 statistical software, the difference of proliferation degree and ratio of each cell lineage in BM smears were compared, the relationship of morphological features of PB smears with BM smears was analyzed. The results showed that obvious or extreme active proliferation of nucleated cells was observed in HAL and NHAL groups, but the myeloproliferation in HAL group was more active than that in NHAL group (P < 0.05). The erythrocyte and megakaryocyte lineages were suppressed in both groups, while the HAL group showed a lower proportion of erythrocyte and megakaryocyte lineages in BM as compared with NHAL group (P < 0.05). The hemoglobin and platelet levels in PB of HAL group were obviously lower than those in PB of NHAL group (P < 0.05). The leukemia cells could be seen in PB smears of NHAL, but the proportion of leukemia cells in NHAL group was smaller than that in HAL group (P < 0.05). The leukocyte count in PB of HAL group strongly positively correlated with the proliferation degree of leukemia cells in BM of HAL group (r = 0.422). It is concluded that the significant difference of proliferation degree, cell levels and blast ratio in BM and PB exists in HAL and NHAL groups, moreover the leukemia cells ratio, leukocyte, hemoglobin and platelet levels in PB of HAL all show characteristic changes. Therefore the contrast analysis of characteristic changes from laboratorial detection contributes to grasp the regular pattern of HAL, meanwhile has an important value for guiding correct diagnosis of acute leukemia and choosing suitable treatment options.