Adult T cell leukemia (ATL) is an aggressive neoplasm of mature helper T cell, which is etiologically linked with human T-lymphotropic virus type 1 (HTLV-I). We studied HTLV-I provirus in 61 cases of ATL with Southern blot analyses and long PCR. These methods detected defective virus in 34 cases (56%). Furthermore, it found two types of defective virus. The first type (type 1) defective virus had both LTRs, but lacked internal sequences, such as gag and pol. Type 1 defective virus was seen in 50% of all defective virus. The second form (type 2) of defective virus had only one LTR, and 5'-LTR was preferentially deleted. This type of defective virus could be more frequently detected in aggressive types of ATL (16/44 cases) than chronic type (1/17 cases). This defective virus might be associated with clinical subtype.