In the fruitfly, Drosophila melanogaster, mate choice during courtship depends on detecting olfactory cues, sex pheromones, which are initially processed in the antennal lobe (AL), a primary olfactory centre of the brain. However, no sexual differences in the structure of the AL have been found in Drosophila. We compared the central brain anatomy of 37 species of Drosophilidae from the islands of the Hawaiian archipelago, uncovering an extreme sexual dimorphism within the AL in which two out of the 51 identifiable glomeruli were markedly enlarged in males. A phylogeny indicated that the sexual dimorphism of the homologous glomeruli arose 0.4-1.9 Myr ago independently in two species groups of Hawaiian endemic Drosophilidae. The corresponding glomeruli in D. melanogaster were also found to be sexually dimorphic. The formation of glomeruli of male size is prevented by the ectopic expression of female-type transformer (tra) cDNA in males, indicating that the glomerular sexual dimorphism is under the control of the sex-determination cascade of genes. It is suggested that a defined set of glomeruli in Drosophila can enlarge in response to sex-determination genetic signals, the mutations of which may result in species differences in sexual dimorphism of the brain.