ObjectivesWe aimed to estimate the burden of neonatal mortality attributable to preterm births associated with maternal HIV infection in sub-Saharan Africa in 1990-2020.DesignModelling study.MethodsEstimates of the excess risk of preterm birth among pregnant women living with HIV (WLHIV) in sub-Saharan Africa in 1990-2020 were obtained from a systematic review and meta-analysis. Data on preterm birth, neonatal mortality, and deaths <1 year directly caused by HIV/AIDS were obtained from the Global Burden of Disease Study 2021. Information on antiretroviral treatment received by WLHIV in sub-Saharan Africa in 1990-2020 were obtained from UNAIDS. These data were used to estimate neonatal mortality due to HIV-attributable preterm birth in countries in sub-Saharan Africa in 1990-2020.ResultsIn 1990-2020, there were an estimated 85,288 (95% confidence interval 53,147-136,921) neonatal deaths due to HIV-attributable preterm births in sub-Saharan Africa. The number of neonatal deaths due to HIV-attributable preterm births in sub-Saharan Africa increased during the 1990 s, before decreasing in the 2000 s, reaching a nadir around 2012, and increasing again within the last decade to levels in 2020 that were similar to their highest levels in the early 2000 s. In 2020, neonatal deaths from HIV-attributable preterm birth exceeded direct HIV/AIDS infant deaths <1 year in sub-Saharan Africa, and by 5-25 fold in countries with a high HIV prevalence.ConclusionsNeonatal deaths due to HIV-attributable preterm birth are increasing and now exceed direct HIV/AIDS infant deaths <1 year in sub-Saharan Africa, particularly in countries with a high HIV prevalence.