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This investigation examined the impact of GWG on infant neurodevelopmental abnormalities at 12\u00a0months of age using the data of a nationwide Japanese cohort study. Questionnaire data were obtained from the ongoing Japan Environment and Children\u2019s Study cohort study. Maternal GWG was subdivided as below, within, or above the reference values of the Institution of Medicine pregnancy weight guidelines. The Ages and Stages Questionnaire, third edition (ASQ-3) is a parent-reported developmental screening instrument for children across five domains: communication, gross motor, fine motor, problem-solving, and personal\u2013social. Multiple logistic regression analysis was employed to identify correlations between GWG and developmental delay defined as ASQ-3 scores of less than two standard deviations below the mean. A total of 30,694 mothers with singleton live births and partners who completed the questionnaire were analyzed. The prevalence of mothers below, within, and above the GWG guidelines was 60.4% (18,527), 32.1% (9850), and 7.5% (2317), respectively. We recorded 10,943 infants (35.7%) who were outliers in at least one ASQ-3 domain. After controlling for covariates, GWG below established guidelines was associated with a significantly higher risk of developmental delay for the communication (odds ratio [OR] 1.21, 95% confidence interval [CI] 1.09\u20131.34), gross motor (OR 1.14, 95% CI 1.05\u20131.24), fine motor (OR 1.13, 95% CI 1.04\u20131.24), problem-solving (OR 1.09, 95% CI 1.01\u20131.18), and personal\u2013social (OR 1.15, 95% CI 1.07\u20131.24) domains.<\/jats:p><jats:p><jats:italic>Conclusion<\/jats:italic>: This large survey revealed a possible deleterious effect of insufficient maternal GWG on infant neurodevelopment.<\/jats:p><jats:p><jats:italic>Trial registration<\/jats:italic>: The Japan Environment and Children\u2019s Study (JECS) was registered in the UMIN Clinical Trials Registry on January 15, 2018 (number UMIN000030786).\n<jats:table-wrap><jats:table><jats:tbody>\n                    <jats:tr>\n                      <jats:td colspan=\"2\"><jats:bold>What is Known:<\/jats:bold><jats:italic>\u2022 Inappropriate maternal gestational weight gain may cause obstetric complications and adverse birth outcomes.<\/jats:italic><jats:italic>\u2022 Excess maternal weight gain may result in gestational diabetes, hypertension, eclampsia, caesarean delivery, and macrosomia, while insufficient maternal weight gain has been associated with pre-term birth and small for gestational age<\/jats:italic>.<\/jats:td>\n                    <\/jats:tr>\n                    <jats:tr>\n                      <jats:td colspan=\"2\"><jats:bold>What is New:<\/jats:bold><jats:italic>\u2022 This study provides important information on a possible adverse effect of insufficient maternal gestational weight gain on offspring neurodevelopment at 12 months of age.<\/jats:italic><jats:italic>\u2022 Our findings indicate a need to reconsider the optimal body mass index and gestational weight gain for women desiring pregnancy.<\/jats:italic><\/jats:td>\n                    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