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The Extraordinary Case of a Woman with a 30-Year-Long Diffuse Leishmaniasis Cured with One Single Ampoule of Intranasal Pentavalent Antimoniate

Sheila V. C. B. Goncalves,Dorcas L. Costa, Joao da J. Cantinho-Junior, Jose N. Vieira-Junior,Edna A. Y. Ishikawa, Rubens N. Costa, Antonio C. G. Costa-Filho, Ronald da C. Araujo,Silvia R. B. Uliana, Jenicer K. U. Y. Yasunaka,Adriano C. Coelho,Jackson M. L. Costa,Carlos H. N. Costa

PATHOGENS(2023)

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Abstract
Infection with Leishmania amazonensis and L. mexicana may lead to diffuse cutaneous leishmaniasis. The cure is exceptional, especially for the strange case of this lady. Case report: The patient acquired the disease in childhood and remained with lesions for over 30 years, albeit several treatments. She worsened after a pregnancy, developing disseminated lesions. Miltefosine with amphotericin B and pentamidine resulted in remission. Lesions reappeared after one year, accompanied by intra-nasal infiltration of the disease. The nasal spraying of a single ampoule of pentavalent antimoniate resulted in the sustained disappearance of the nasal symptoms and all the cutaneous lesions. After over eight years, she remains disease-free, albeit under renal replacement therapy. The high nasal mucosal antimonial concentration may explain the long-lasting cure via new MHC class I epitope-specific CD8+ cell clones against L. amazonensis present in the nasal mucosa.
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Key words
diffuse cutaneous leishmaniasis,pentavalent antimonial,intranasal drug delivery,CD8+ T-cells,anergy,immune cell exhaustion
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