Introduction Mucosal leishmaniasis (ML) is a rare, destructive, and recurrent protozoal infection caused by Leishmania species, most commonly Leishmania braziliensis . ML can develop months to years after initial cutaneous leishmaniasis (CL) and may lead to severe mucosal destruction if untreated. Case Presentation We present a case of a 48‐year‐old healthy male from Venezuela with prior CL treated with intravenous meglumine antimoniate who subsequently developed progressive nasal symptoms consistent with ML. Initial therapy with oral fluconazole was ineffective, but treatment with miltefosine led to symptom resolution. The patient underwent four‐staged reconstructive surgeries with ENT. Thirty‐eight months later, the patient developed recurrent symptoms, and biopsy confirmed infection with L. braziliensis . He was subsequently treated with intravenous amphotericin B, which led to clinical improvement. Discussion This case highlights the relapsing nature of ML, its potential for extensive mucosal damage, and the importance of a structured, long‐term care plan. This case in particular is unique given the extended delay before recurrence following miltefosine therapy. The high recurrence rate of L. braziliensis infections underscores the importance of vigilant long‐term follow‐up and consideration of systemic therapy. Multidisciplinary management is often necessary to optimize outcomes. Conclusion Early recognition of ML, timely systemic therapy, structured multidisciplinary care, and adherence to a long‐term surveillance plan are critical to prevent mucosal destruction and manage relapses effectively. Extended surveillance may be warranted to detect late relapses in patients treated with newer or more effective agents.
Ehsan et al. (2026) studied this question.