The rapid spread of dengue fever (DF) infection has posed severe threats to global health. Environmental factors, such as weather conditions, are believed to regulate DF spread. While previous research reported inconsistent change of DF risk with varying weather conditions, few of them evaluated the impact of extreme weather conditions on DF infection risk. This study aims to examine the short-term associations between extreme temperatures, extreme rainfall, and DF infection risk in South and Southeast Asia. A total of 35 locations in Singapore, Malaysia, Sri Lanka, and Thailand were included, and weekly DF data, as well as the daily meteorological data from 2012 to 2020 were collected. A two-stage meta-analysis was used to estimate the overall effect of extreme weather conditions on the DF infection risk. Location-specific associations were obtained by the distributed lag nonlinear models. The DF infection risk appeared to increase within 1-3 weeks after extremely high temperature (e.g. lag week 2: RR = 1.074, 95 % CI: 1.022-1.129, p = 0.005). Compared with no rainfall, extreme rainfall was associated with a declined DF risk (RR = 0.748, 95 % CI: 0.620-0.903, p = 0.003), and most of the impact was across 0-3 weeks lag. In addition, the DF risk was found to be associated with more intensive extreme weathers (e.g. seven extreme rainfall days per week: RR = 0.338, 95 % CI: 0.120-0.947, p = 0.039). This study provides more evidence in support of the impact of extreme weather conditions on DF infection and suggests better preparation of DF control measures according to climate change.