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Thrombocytopenia in surgical patients is a potentially serious condition, faced by surgeons. A close relationship between sepsis and thrombocytopenia has been suggested. Thrombocytopenia has even been suggested to be indicative of an acute infection. Platelet count in a septicemic patient may also serve as a prognostic tool. There are many reports of thrombocytopenia due to septicemia in the literature but the occurrence of thrombocytopenia in maxillofacial infections is rare. Thrombocytopenia in a patient with odontogenic infection presents unique diagnostic and management challenges. A case report of an adult male patient with odontogenic infection, who developed life-threatening thrombocytopenia, is presented.