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Introduction and Objectives: Hypospadias is a congenital disorder in boys in which the urethral meatus is located on the ventral penis. The incidence rate of hypospadias is increasing, however, there is still no literature regarding how significant is the role of maternal and environmental factors to the risk of hypospadias, especially in Indonesia. This study aims to analyze the maternal and environmental factors during pregnancy to the risk of hypospadias., Materials and Methods: This study was an analytical observational study with a case control design. The subjects were mothers who gave birth to children with hypospadias, compared to normal, who visited the urology, pediatric-surgery, plastic-surgery, and pediatric clinic at Hasan Sadikin General Hospital., Results: A total of 120 samples (60 cases and 60 control) were included in this study. There were 14 maternal factors, which 5 of them found significantly correlated with hypospadias, including maternal occupation as an industrial worker ( P  = .003; OR:4.789), pregnancy-enhancing drugs usage ( P  = .004; OR: 5.783), smoking consumption ( P  = .034; OR: 2.294), mosquito-repellant usage ( P  = .0001; OR: 82.600), and preterm birth ( P  = .013; OR: 2.895). There were 2 environmental factors, and one of them was significant, which was the distance from home to industrial/rice fields/waste areas, approximately 780 m ( P  = .0001; OR: 6.102). Based on multivariate analysis, we found that maternal occupation, mosquito repellent usage, and the distance from home to industrial area had a strong relationship in predicting hypospadias occurrence., Conclusion: Several maternal and environmental factors were found significantly correlated to hypospadias occurrence. Among these, mosquito-repellant usage was found to be the most significant factor., Competing Interests: Declaration of Conflicting Interests: The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article., (© The Author(s) 2022.)