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Due to the sparse data on benzene exposure and myelodysplastic syndrome (MDS) subtypes, we studied this relationship in patients from 29 hospitals in Shanghai, China.We recruited 604 cases of MDS and 1193 controls matched on age, sex, and admission date. We interviewed subjects for information on workplace and lifestyle exposures, and developed semi-quantitative exposure estimates.Benzene exposure showed a direct exposure–response pattern with refractory cytopenia with multilineage dysplasia, a less certain association with refractory cytopenia with unilineage dysplasia, and no association with other MDS subtypes. A different pattern was observed with farm residence and smoking, which was primarily related to refractory anemias.This research demonstrates the importance of MDS subtype specification for more robust etiologic insights. Our data suggests that subtypes with non-erythroid dysplasia are associated with benzene exposure.