Background: Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening, hyperinflammatory syndrome, characterized by the uncontrolled activation of macrophages and T cells, eliciting key symptoms such as persistent fever, hepatosplenomegaly, pancytopenia, hemophagocytosis, hyperferritinemia, and coagulopathy. Viral infections are frequently implicated in the onset of active HLH episodes, both in primary, genetic HLH as in the secondary, acquired form. Despite that, and a large epidemics of Chikungunya (CHIKV) no cases have been associated with this arbovirus. Methods & Materials: Case report and systematic review of literature. Results: A 42-year-old man consult for 15 days of fever, rash and evanescent symmetrical polyarthralgias predominantly in trunk and upper limbs and hepatosplenomegaly. Laboratory tests evidence thrombocytopenia and anemia without leukopenia, hyperbilirubinemia (indirect fraction), elevated ESR, high ferritin, positive serology (IgG/IgM) for CHIKV. A bone marrow aspirate showed lymphohistiocytosis and haemophagocytosis. Other infectious diseases, were ruled out. Conclusion: HLH, can be familiar or acquired, it is rare cause of fever of unknown origin. Given the growing epidemic of arboviral infections, including CHIKV, it is necessary to describe the finding of hemophagocytic lymphohistiocytosis, triggered in this case by such alphavirus infection. HLH is a multisystem condition in this case associated with CHIKV infection. Given the context of CHIKV as endemic disease in Colombia and other countries in Latin America, other similar cases can occur.