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Tongue Nodule as Primary Manifestation of American Cutaneous Leishmaniasis in an Immunocompetent Patient

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Abstract

Leishmaniasis is a parasitic disease considered an endemic public health problem in developing countries, where it is a reportable disease. Isolated oral manifestation is rare, and its clinical manifestations are variable. In this paper we describe an unusual case of an immunocompetent patient, 57-year-old man with a painless reddish submucosal nodule located on the tongue dorsum. Microscopical analysis showed chronic inflammatory infiltrate with macrophages containing leishmania in cytoplasmic vacuoles. PCR assays confirmed the diagnosis and patient was treated with meglumine antimoniate for 30 days. Absence of the parasite was confirmed by PCR. Thirteen years after treatment, a scar fibrosis persisted on the tongue dorsum. The case reported reveals that leishmaniasis should be considered in the diagnosis of tongue nodules in immunocompetent patients.

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References

  1. Okwor I, Uzonna J. Social and economic burden of human leishmaniasis. Am J Trop Med Hyg. 2016;94:489–93. https://doi.org/10.4269/ajtmh.15-0408.

    Article  PubMed  PubMed Central  Google Scholar 

  2. Santos CR, Tuon FF, Cieslinski J, de Souza RM, Imamura R, Amato VS. Comparative study on liposomal amphotericin B and other therapies in the treatment of mucosal leishmaniasis:A15-yearretrospectivecohortstudy. PLoS ONE. 2018a;14:23–31. https://doi.org/10.1371/journal.pone.0218786.

    Article  CAS  Google Scholar 

  3. Daneshbod Y, Oryan A, Davarmanesh M, Shirian S, Negahban S, Aledavood A, et al. Clinical, histopathologic, and cytologic diagnosis of mucosal leishmaniasis and literature review. Arch Pathol Lab Med. 2011;135:478–82. https://doi.org/10.1043/2010-0069-OA.1.

    Article  PubMed  Google Scholar 

  4. Motta ACF, Lopes MA, Ito FA, Carlos-Bregni R, De Almeida OP, Roselino AM. Oral leishmaniasis: a clinicopathological study of 11 cases. Oral Dis. 2007;13:335–40. https://doi.org/10.1111/j.1601-0825.2006.01296.x.

    Article  CAS  PubMed  Google Scholar 

  5. Almeida TFA, da Silveira EM, dos Santos CRR, León JE, Mesquita ATM. Exclusive primary lesion of oral leishmaniasis with immunohistochemical diagnosis. Head Neck Pathol. 2016;10:533–7. https://doi.org/10.1007/s12105-016-0732-7.

    Article  PubMed  PubMed Central  Google Scholar 

  6. World Health Organization. Leishmaniasis. World Health Org Fact Sheet. World Heal. Organ. 2020. http://www.who.int/mediacentre/factsheets/fs375/en/.

  7. Pan American Health Organization. Leishmaniasis: Epidemiological Report in the Americas. Washington, DC: PAHO. 2019. p. 2–5. http://iris.paho.org/xmlui/handle/123456789/50505.

  8. Brazil MS. Tegumentary Leishmaniasis (TL): what it is, causes, symptoms, treatment, diagnosis and prevention. Tegumentary Leishmaniasis what it is, causes, symptoms, Treat diagnosis Prev. 2019. p. 4. http://www.saude.gov.br/saude-de-a-z/leishmaniose-tegumentar.

  9. Akhoundi M, Kuhls K, Cannet A, Votýpka J, Marty P, Delaunay P, et al. A historical overview of the classification, evolution, and dispersion of leishmania parasites and sandflies. PLoS Negl Trop Dis. 2016. https://doi.org/10.1371/journal.pntd.0004349.

    Article  PubMed  PubMed Central  Google Scholar 

  10. Handler MZ, Patel PA, Kapila R, Al-Qubati Y, Schwartz RA. Cutaneous and mucocutaneous leishmaniasis: clinical perspectives. J Am Acad Dermatol. 2015;73:897–908. https://doi.org/10.1016/j.jaad.2014.08.051.

    Article  PubMed  Google Scholar 

  11. Goto H, Lauletta Lindoso JA. Cutaneous and mucocutaneous leishmaniasis. Infect Dis Clin N Am. 2012;26:293–307. https://doi.org/10.1016/j.idc.2012.03.001.

    Article  Google Scholar 

  12. Nadler C, Enk CD, Leon GT, Samuni Y, Maly A, Czerninski R. Diagnosis and management of oral leishmaniasis—case series and literature review. J Oral Maxillofac Surg. 2014;72:927–34. https://doi.org/10.1016/j.joms.2013.10.021.

    Article  PubMed  Google Scholar 

  13. Teemul TA, Giles-Lima M, Williams J, Lester SE. Laryngeal leishmaniasis: case report of a rare infection. Head Neck. 2013. https://doi.org/10.1002/hed.23141.

    Article  PubMed  Google Scholar 

  14. da Costa DC, Palmeiro MRMJ. Oral manifestations in the American tegumentary leishmaniasis. PLoS ONE. 2015. https://doi.org/10.1371/journal.pone.0117681.

    Article  PubMed  PubMed Central  Google Scholar 

  15. Garg S, Tripathi R, Tripathi K. Oral mucosal involvement in visceral leishmaniasis. Asian Pac J Trop Med. 2013;6:249–50. https://doi.org/10.1016/S1995-7645(13)60034-6.

    Article  PubMed  Google Scholar 

  16. Palmeiro MR, Rosalino CMV, Quintella LP, Morgado FN, da Costa Martins AC, Moreira J, et al. Gingival leishmaniasis in an HIV-negative patient. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol. 2007. https://doi.org/10.1016/j.tripleo.2007.07.008.

    Article  Google Scholar 

  17. Marsden PD. Mucosal leishmaniasis (“espundia” escomel, 1911). Trans R Soc Trop Med Hyg. 1986;80:859–76. https://doi.org/10.1016/00359203(86)90243-9.

    Article  CAS  PubMed  Google Scholar 

  18. Strazzulla A, Cocuzza S, Pinzone MR, Postorino MC, Cosentino S, Serra A, et al. Mucosal leishmaniasis: an underestimated presentation of a neglected disease. Biomed Res Int. 2013. https://doi.org/10.1155/2013/805108.

    Article  PubMed  PubMed Central  Google Scholar 

  19. Sánchez-Romero C, Júnior HM, da Matta VLR, Freitas LM, Soares CM, Mariano FV, et al. Immunohistochemical and molecular diagnosis of mucocutaneous and mucosal leishmaniasis. Int J Surg Pathol. 2020;28:138–45. https://doi.org/10.1177/1066896919876706.

    Article  CAS  PubMed  Google Scholar 

  20. Deepachandi B, Weerasinghe S, Soysa P, Karunaweera N, Siriwardana Y. A highly sensitive modified nested PCR to enhance case detection in leishmaniasis. BMC Infect Dis. 2019. https://doi.org/10.1186/s12879-019-4180-3.

    Article  PubMed  PubMed Central  Google Scholar 

  21. Mignogna MD, Celentano A, Leuci S, Cascone M, Adamo D, Ruoppo E, et al. Mucosal leishmaniasis with primary oral involvement: a case series and a review of the literature. Oral Dis. 2015;21:e70–8. https://doi.org/10.1111/odi.12268.

    Article  CAS  PubMed  Google Scholar 

  22. Bogdan C. Leishmaniasis in rheumatology, haematology and oncology: epidemiological, immunological and clinical aspects and caveats. Ann Rheum Dis. 2012. https://doi.org/10.1136/annrheumdis-2011-200596.

    Article  PubMed  Google Scholar 

  23. Akuffo H, Costa C, van Griensven J, Burza S, Moreno J, Herrero M. Newinsights into leishmaniasis in the immunosuppressed. PLoS Negl Trop Dis. 2018. https://doi.org/10.1371/journal.pntd.0006375.

    Article  PubMed  PubMed Central  Google Scholar 

  24. Garcíade Marcos JA, Dean Ferrer A, Alamillos Granados F, Ruiz Masera JJ, Cortés Rodríguez B, Vidal Jiménez A, et al. Localized leishmaniasis of the oral mucosa. A report of three cases. Med Oral Patol Oral Cir Bucal. 2007. https://doi.org/10.1371/journal.pntd.0006375.

    Article  Google Scholar 

  25. Pellicioli ACA, Martins MAT, Filho MSA, Rados PV, Martins MD. Leishmaniasis with oral mucosa involvement. Gerodontology. 2012. https://doi.org/10.1111/j.1741-2358.2011.00512.x.

    Article  PubMed  Google Scholar 

  26. Vicente CR, Falqueto A. Differentiation of mucosal lesions in mucocutaneous leishmaniasis and paracoccidioidomycosis. PLoS ONE. 2018. https://doi.org/10.1371/journal.pone.0208208.

    Article  PubMed  PubMed Central  Google Scholar 

  27. de Arruda JAA, Schuch LF, Abreu LG, Silva LVO, Mosconi C, Monteiro JLGC, et al. A multicentre study of oral paracoccidioidomycosis: analysis of 320 cases and literature review. Oral Dis. 2018;24:1492–502. https://doi.org/10.1111/odi.12925.

    Article  PubMed  Google Scholar 

  28. Mazumder SA, Pandey S, Brewer SC, Baselski VS, Weina PJ, Land MA, et al. Lingual leishmaniasis complicating visceral disease. J Travel Med. 2010;17:212–4. https://doi.org/10.1111/j.1708-8305.2010.00403.x.

    Article  PubMed  Google Scholar 

  29. Amato VS, Tuon FF, Bacha HA, Neto VA, Nicodemo AC. Mucosal leishmaniasis. Current scenario and prospects for treatment. Acta Trop. 2008;105:1–9. https://doi.org/10.1016/j.actatropica.2007.08.003.

    Article  CAS  PubMed  Google Scholar 

  30. World Health Organization/Pan American Health Organization. Leishmaniasis in the Americas: treatment recommendation. 2018. p. 60. http://iris.paho.org/xmlui/bitstream/handle/123456789/7704/9789275117521_eng.pdf?sequence=5&isAllowed=y.

  31. Santos CR, Tuon FF, Cieslinski J, de Souza RM, Imamura R, Amato VS. Comparative study on liposomal amphotericin B and other therapies in the treatment of mucosal leishmaniasis: a 15-year retrospective cohort study. PLoS ONE. 2018b. https://doi.org/10.1371/journal.pone.0218786.

    Article  PubMed  PubMed Central  Google Scholar 

  32. Brazil. Ministry of Health. Secretariat of Health Surveillance. Department of Surveillance of Communicable Diseases. Tegumentary leishmaniasis surveillance manual. 2017. p. 191. http://www.dive.sc.gov.br/conteudos/publicacoes/17_0093_M_e_C.pdf.

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Acknowledgements

We thank Dra Lucile Maria Floeter-Winter from Department of Physiology, Institute of Biosciences, University of São Paulo (USP), for performing the PCR assays.

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Correspondence to Daniel Galera Bernabé.

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Botelho, M.C.B., Ferreira, L.L., Fikaris, S. et al. Tongue Nodule as Primary Manifestation of American Cutaneous Leishmaniasis in an Immunocompetent Patient. Head and Neck Pathol 15, 1069–1073 (2021). https://doi.org/10.1007/s12105-020-01253-w

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