Natural aryltetralin lactone lignans existed in the plants of family Berberidaceae, Acanthaceae, Burseraceae, Verbenaceae, Euphorbiaceae, etc. Due to the antineoplastic and antiviral properties, it has become a hot research topic in medicinal chemistry. This review covers extraction and isolation, biosynthesis, plant origin, and structure and spectral characteristics of natural aryltetralin lactone lignans. It will provide a useful reference for the intensive studies and rational utilization of aryltetralin lactone lignans.