Neural tube defect (NTD) is not uncommon in Bangladesh. Among the nutritional factors related to neural tube defects, in addition to Folate, low level of serum zinc has been described by many authors in the recent time. This study was conducted with the objective of finding the relationship of serum zinc with NTD in a Bangladeshi population. This case control study was conducted between in BSMMU, Dhaka. Newborns with neural tube defects and their mothers were considered as cases and normal babies and their mothers were taken as controls. Mothers taking zinc supplements in any part of pregnancy were excluded from the study. Venous blood specimens from mothers and newborns were obtained at or within one week of delivery. Serum zinc concentration was determined by Atomic Absorption Spectrophotometry. Low level of serum zinc in the mothers and newborns were found to be associated with neural tube defects, which is consistent with some other studies. However, it is also evident that some other factors (maternal parity etc) could significantly be associated with this defect. To confirm these findings, a further study with a larger sample size is recommended.