Aepyosciurinae, a new subfamily of Sciuridae, were found at the base of the early Pleistocene loess deposits in Dongxiang County, Gansu Province. Its unilaterally hypsodont and lophodont cheek teeth are unique among the sciurids so far known all over the world. Certain degree of similarity can be observed between the cheek teeth of the new subfamily and the Anomalurinae living in tropical and subtropical forests in central and western Africa. Aepyosciuris orientalis gen. et sp. nov. might have lived in montaneous woodland or grassland and lived on harder leaves, barks, or even grass. This tends to show that the northeastern border area of the Tibetan Plateau had been lifted considerably high in early Pleistocene (ca. 2 Ma), with drier climate, becoming a suitable habitat for Aepyosciurus orientalis.