: As hepatitis C elimination efforts are launched, national strategies for screening and treatment scale-up in countries, such as Malaysia, must be designed and implemented. Strategic information, including estimates of the total number of patients chronically-infected with hepatitis C virus (HCV) and the size of key populations, such as people who inject drugs (PWID), is critical to informing these efforts. For Malaysia, the estimate of the PWID population size most frequently reported in global systematic reviews is for the year 2009. : To support ongoing national HCV planning efforts, we aimed to estimate the national population size of active PWID in Malaysia, for the years 2014 and 2017. : To estimate the PWID population size, we applied standard benchmark-multiplier methodology, frequently used for PWID population size estimation, and extended it by adjusting for cessation of injecting drug use within the benchmark and calculating statistical uncertainty intervals. : The estimated active PWID population size was 153,000 (95% uncertainty interval (UI): 136,000-172,000) for 2014 and 156,000 (95% UI: 137,000-188,000) for 2017. /: This updated estimate of the active PWID population size in Malaysia will help inform effective planning for the scale-up of HCV screening and treatment services. The proposed methodology is applicable to other countries that maintain national HIV registries and have conducted Integrated Biological and Behavioral Surveys among active PWID.