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We present a case of endonasal leishmaniasis diagnosed on a frozen section biopsy specimen obtained from a 47-year-old woman treated with corticosteroids and anti-TNF^α for rheumatoid arthritis. Similar cases of isolated mucosal leishmaniasis have not yet been reported in Croatia. Microscopic analysis of the biopsy specimen sent for frozen sections revealed Leishmania amastigotes in macrophages infiltrating nasal mucosa. Subsequent communication with the referring clinician revealed that the patient had been diagnosed with visceral leishmaniasis 13 years prior to current biopsy. Thus, the present nasal lesion was interpreted as a solitary lesion related to reactivation of the previous infection. She received liposomal amphotericin B which led to prompt amelioration of the lesion. As shown in the present case, clinical presentation of leishmaniasis is unspecific and can mimic many other conditions, especially in the setting of concomitant immunosuppressive and anti-inflammatory treatment. It is important to be aware of this entity in order to treat such patients properly.