Manganese (Mn) is an essential mineral nutrient in mammals. The physiological role of Mn in animal models is well documented, but little is known about the adverse effects of Mn deficiency or overexposure in humans, including pregnancy outcomes such as birth weight. We examined the relationship of the maternal and cord blood Mn levels with birth weight in a cohort of 172 mother-infant pairs born in Shanghai, China. Non-linear spline and quadratic regression models were used to test the hypothesis of an inverted U-shaped association between the Mn levels and birth weight. The median (range) levels of Mn in the maternal and cord blood were 5.38 (2.34-30.37) μg/dL and 7.66 (2.57-34.23) μg/dL, respectively. An inverted U-shaped relationship was observed between maternal Mn and birth weight after adjusting for potential confounders. The birth weight increased with Mn levels up to 4.18 μg/dL, and a slight reduction in weight was observed at higher levels. The cord blood Mn levels were not found to be associated with birth weight. Both lower and higher Mn exposures are associated with lower birth weight, which may influence important developmental parameters; the association of higher Mn levels with lower weight was weak and imprecise.