The first human isolate of Borrelia lusitaniae recovered from a Portuguese patient with suspected Lyme borreliosis is described. This isolate, from a chronic skin lesion, is also the first human isolate of Borrelia in Portugal. Different phenotypic and molecular methods are used to characterize it

This culture confirmed skin infection by B. lusitaniae in a patient from Portugal suggests an additional human pathogen out of the B. burgdorferi sensu lato complex in Europe, particularly in Portugal.