BackgroundThe presence of aluminium (Al) in the human body may impact brain neurodevelopment and function, and it is thought to contribute to autism spectrum disease (ASD). The main objective of this study was to assess the association between urinary Al and the development of ASD among Malaysian preschool children in the urban city of Kuala Lumpur.MethodThis was an unmatched case–control study in which children with ASD were recruited from an autism early intervention center and typically developed (TD) children were recruited from government-run nurseries and preschools. Urine samples were collected at home, assembled temporarily at study locations, and transported to the laboratory within 24 h. The Al concentration in the children’s urine samples was determined using inductively coupled plasma mass spectrometry (ICP-MS).ResultA total of 155 preschool children; 81 ASD children and 74 TD children, aged 3 to 6 years, were enlisted in the study. This study demonstrated that ASD children had significantly higher urinary Al levels than TD children (median (interquartile range (IQR): 2.89 (6.77) µg/dL versus 0.96 (2.95) µg/dL) (p< 0.001). Higher parental education level, non-Malay ethnicity, male gender, and higher urinary Al level were the significant ASD risk factors (adjusted odds ratio (aOR) >1,p< 0.05).ConclusionA higher urine Al level was discovered to be a significant risk factor for ASD among preschool children in the urban area of Kuala Lumpur, Malaysia.