Non-perennial rivers are valuable water resources that support millions of humans globally, as well as unique riparian ecosystems. In Australia, the Earth's driest inhabited continent, over 70% of rivers are non-perennial due to a combination of ancient landscape, dry climates, highly variable rainfall regimes, and human interventions that have altered riverine environments. Here, we review Australian non-perennial river research incorporating geomorphology, hydrology, biogeochemistry, ecology, and Indigenous knowledges. The dominant research themes in Australia were drought, floods, salinity, dryland ecology, and water management. Future research will likely follow these themes but must address emerging threats to river systems due to climate change and other anthropogenic impacts. Four high level opportunities for future research are identified, namely: (1) integrating Indigenous and western scientific knowledge; (2) quantifying climate change impacts on hydrological and biological function; (3) clarifying the meaning and measurement of “restoration” of non-perennial systems; and (4) understanding the role of groundwater. These challenges will require inter- and multi-disciplinary efforts supported by technological advances. The evolving body of knowledge about Australian rivers provides a foundation for comparison with other dryland areas globally where recognition of the importance of non-perennial rivers is expanding.