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Objective: To report a case of posaconazole induced diffuse lentigines. Case presentation: A 57-year-old Thai male with acute promyelocytic leukemia who received chemotherapy and antifungal drug prophylaxis then appeared abruptly generalized multiple discrete well-defined brownish macules on face, lips, trunk, back, both arms, forearms, both thighs, legs, both palms and soles about eight days after received posaconazole. Conclusion: Multiple lentigines could be found in patients who received immunosuppression or immunomodulation, and antifungal drugs such as voriconazole.