Plasmodium knowlesi is now added to the known four Plasmodium species (P. vivax, P. falciparum, P. malariae, P. ovale) as a cause of malaria in humans because of the recent increasing rate of cases reported from countries of southeastern Asia. P. knowlesi which infects macaque monkeys (Macaca fascicularis and M. nemestrina) is transmitted to humans especially by Anopheles leucosphyrus and An. hackeri mosquitos. First human cases of P. knowlesi malaria have been detected in Malaysia which have reached high numbers in recent years and also have been reported from countries of Southeast Asia such as Thailand, Philippines, Myanmar, Singapore and Vietnam. However the number of cases reported from western countries are rare and limited only within voyagers. This report is the first presentation of an imported case of P. knowlesi malaria in Turkey and aims to draw attention to the point that it could also be detected in future. A 33-year-old male patient from Myanmar who has migrated to Turkey as a refugee, was admitted to a health center with the complaints of fever with a periodicity of 24 hours, headache, fatigue, cough, sore throat, anorexia, myalgia and arthralgia. He was prediagnosed as upper respiratory tract infection, however because of his periodical fever and background in Myanmar, thick and thin blood films were prepared and sent to our laboratory for further examinations. Microscopic examination of the thin blood films revealed erythrocytic stages compatible with P. knowlesi (three large early trophozoites in an erythrocyte, three late trophozoites with compact view, and three late band-form trophozoites). Upon this, both real-time polymerase chain reaction (Rt-PCR) targeting the small subunit ribosomal RNA (SSU-rRNA) genes of Plasmodium genus and DNA sequence analysis targeting P. knowlesi rRNA gene were performed. As a result, the suspected identification of P. knowlesi by microscopy was confirmed by Rt-PCR and DNA sequencing. The patient was treated with chloroquine and primaquine combination and in the follow-up on the seventh day after the treatment, his parasitemia and symptoms had ceased. Although there were some previous reports concerning about imported patients infected with different Plasmodium species in our country, no cases of P. knowlesi have been reported. This first case presented here emphasizes the occurence of P. knowlesi malaria in Turkey hereinafter due to the increasing number of refugees.