Galls are remarkable parasite–plant interactions that develop in different organs. They are induced by various organisms which manipulate or reprogram plant development. Galls in dioecious species and their effects on the host plant are seldom described in the literature. This paper presents a novel study of galls in a dioecious plant of the neotropical region. Our study aimed to analyze gall development and describe morphological and metabolic changes in galled leaves caused by Cecidomyiidae (Diptera) in Clusia fluminensis (Clusiaceae), a dioecious plant from Brazilian restinga. We investigated male and female individuals to detect different responses and sex-biased interactions. The non-galled leaves of female and male individuals of C. fluminensis exhibited similar anatomical structures. Nevertheless, galls developed only in male individuals. The activity of the Cecidomyiidae led to several morphological and anatomical changes, such as the hypertrophy of the leaf blade, especially the chlorenchyma. Our results indicated an interesting exception for the pattern of storage of lipids and starch in Cecidomyiidae galls, and sex-biased development in a dioecious plant, with the variation of metabolic compounds, especially phenolics and flavonoids, which may inhibit gall development in female individuals.