The Amazon rainforest and the Brazilian Cerrado both possess high phlebotomine diversity. The fragmentation of these habitats has resulted in the appearance of human cases of cutaneous leishmaniasis. In one altered area of mixed primary vegetation (forest and Cerrado) and its adjacent settlement in the northeast state of Maranhão, Brazil, evidence exists for the active transmission of cutaneous leishmaniasis. Accordingly, an entomological investigation was performed in both the forest and the settlement to compare the phlebotomine vector fauna in each environment. The study was conducted from September 2009 to August 2010 in the municipality of Itapecuru Mirim in the state of Maranhão, Brazil. The phlebotomine species were captured using 24 light Center for Disease Control and Prevention traps that were placed in the forest and the settlement (peridomicile and intradomicile). The similarity between the phlebotomine compositions in the forest and those in the settlement was determined using a Principal Coordinate Analysis based on a dissimilarity matrix that was calculated using the Bray—Curtis index (relative abundance) and the Jaccard index (presence and absence of species). In total, 29 Lutzomyia species and one Brumptomyia species were collected. The phlebotomines were diverse and abundant in both the forest fragment (27 species, 4,606 specimens) and the settlement (22 species, 753 specimens). The most abundant species were L. infraspinosa (25%), L. davisi (21%), L. antunesi (21%), L. longipalpis (9%), L. saulensis (6%), L. flaviscutellata (5%), and L. wellcomei (4%). Some species were found strictly in the forest, other species were exclusive to the anthropic environment, and some species colonized both of the studied environments. The phlebotomines adaptation to these modified environments explains the autochthonous outbreak of cutaneous leishmaniasis.