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Drug-induced thrombocytosis is difficult to identify and is rarely reported. Here, we report a probable rare case of imipenem–cilastatin-induced thrombocytosis in a 51-year-old female. The patient received imipenem–cilastatin treatment because of an intra-abdominal infection, following which the platelet count increased to a peak value of 1058×10 9 cells/L in 14 days. When imipenem–cilastatin was replaced by levofloxacin and amikacin, the platelet count decreased progressively and remained within the normal range until the 6-month follow-up. Moreover, platelet counts were independent of the infection. Thrombocytosis was probably attributable to imipenem–cilastatin with a Naranjo score of seven. This is a probable rare case in which the thrombocytosis was likely an adverse reaction induced by imipenem–cilastatin. We suggest that clinicians need to be vigilant concerning the impact of imipenem–cilastatin on platelet count in patients with thrombocytosis.