High brucellosis seroprevalence rates in domestic swine herds have been reported in Wallis and Futuna Islands and are associated with a significant burden of human infection by Brucella suis, a species that is rarely incriminated in human disease. Between 2003 and 2010, seven patients had a positive blood culture for B. suis biovar 1, 11 symptomatic patients had a positive Rose Bengal test (RBT) and a positive serum agglutination test (SAT) and three asymptomatic cases were found to be positive for RBT, SAT or ELISA IgM (after systematic screening of 52 family members of 15 index cases). Overall, Brucella infection was diagnosed in 21 people, corresponding to a mean annual incidence of 19 cases/100 000 inhabitants. Compared to series of patients infected with other more commonly encountered Brucella spp. such as B. melitensis and B. abortus, clinical presentation and percentage and distribution of complications were similar, apart from a marked observation of significantly increased median alanine aminotransferase levels, 20 times greater than upper normal rates, but not accompanied by any particular hepatic pathology. Wallis and Futuna, where people live in close proximity to animals and where the cultural significance of pig-raising precludes the implementation of adequate veterinary preventive measures, thus represents one of the few known B. suis foci worldwide and allows for evaluation of the peculiarities of this infection.