The first confirmed case of Rickettsia parkeri infection in Uruguay is reported. To date, in South America, molecularly confirmed cases of human infection have been found in Argentina and probably, Brazil. Our patient returned to Spain after a 7-day trip to Colonia Suiza (Southwestern Uruguay). He presented fever (39°C), chills, and two eschars (tache noire-like) surrounded by an indurated, erythematous halo on the inner side of the left ankle besides a maculopapular rash on the legs. After treatment with doxycycline for 7 days, he fully recovered. R. parkeri infection was diagnosed by molecular-based detection of the microorganism in a swab specimen of the eschar. Diagnosis was supported by seroconversion between acute- and convalescent-phase sera specimens.