Leishmaniasis is a protozoal infection transmittedby a sandfly vector. In Germany, leishmaniasis ofthe mucous membranes is a rare condition andusually due to extension of local skin disease intothe mucosal tissue via direct extension,bloodstream or lymphatics. We report a case ofendonasal leishmaniasis in a female Germanresident who presented in a university hospitalwith nasal obstruction. Histology of the left nasalseptum biopsy was suggestive of leishmaniasis.The molecular detection of DNA was positive forleishmania infantum. The patient was successfullytreated as a case of mucocutaneous leishmaniasisreceiving liposomal amphotericin follow up visitsshowed significant improvement with norecurrence

Abrar Adnan Suqati

Annett Pudszuhn

Veit Maria Hofmann

English

AJOL - African Journals Online

Article    Abrar Adnan Suqati et al. PAMJ - 36(292). 17 Aug 2020.  -  Page numbers not for citation purposes. 1 Case report  Mucocutaneous leishmaniasis: case report and literature review of a rare endonasal infection  Abrar Adnan Suqati, Annett Pudszuhn, Veit Maria Hofmann Corresponding author: Abrar Adnan Suqati, Department of Otorhinolaryngology, Head and Neck Surgery, Charité-Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin, Humboldt-Universität zu Berlin and Berlin Institute of Health, Campus Benjamin Franklin, Berlin, Germany. abrar.suqati@charite.de Received: 24 Jun 2020  -  Accepted: 30 Jun 2020  -  Published: 17 Aug 2020 Keywords: Cutaneous infection, leishmaniasis, traveler’s diseases  Copyright: Abrar Adnan Suqati et al. Pan African Medical Journal (ISSN: 1937-8688). This is an Open Access article distributed under the terms of the Creative Commons Attribution International 4.0 License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Cite this article: Abrar Adnan Suqati et al. Mucocutaneous leishmaniasis: case report and literature review of a rare endonasal infection. Pan African Medical Journal. 2020;36(292). 10.11604/pamj.2020.36.292.24543 Available online at: https://www.panafrican-med-journal.com/content/article/36/292/full  Mucocutaneous leishmaniasis: case report and literature review of a rare endonasal infection Abrar Adnan Suqati1,&, Annett Pudszuhn1, Veit Maria Hofmann1 1Department of Otorhinolaryngology, Head and Neck Surgery, Charité-Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin, Humboldt-Universität zu Berlin and Berlin Institute of Health, Campus Benjamin Franklin, Berlin, Germany &Corresponding author Abrar Adnan Suqati, Department of Otorhinolaryngology, Head and Neck Surgery, Charité-Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin, Humboldt-Universität zu Berlin and Berlin Institute of Health, Campus Benjamin Franklin, Berlin, Germany     Article    Abrar Adnan Suqati et al. PAMJ - 36(292). 17 Aug 2020.  -  Page numbers not for citation purposes. 2 Abstract Leishmaniasis is a protozoal infection transmitted by a sandfly vector. In Germany, leishmaniasis of the mucous membranes is a rare condition and usually due to extension of local skin disease into the mucosal tissue via direct extension, bloodstream or lymphatics. We report a case of endonasal leishmaniasis in a female German resident who presented in a university hospital with nasal obstruction. Histology of the left nasal septum biopsy was suggestive of leishmaniasis. The molecular detection of DNA was positive for leishmania infantum. The patient was successfully treated as a case of mucocutaneous leishmaniasis receiving liposomal amphotericin follow up visits showed significant improvement with no recurrence. Introduction     Leishmaniasis is a protozoal infection transmitted by a sandfly vector. According to the World Health Organization (WHO), mucocutaneous leishmaniasis (MCL) is endemic in Central and South America. Almost 90% of MCL cases occur in Bolivia, Brazil and Peru [1]. In Germany leishmaniasis of the mucous membranes is a rare condition and usually due to extension of local skin disease into the mucosal tissue via direct extension, bloodstream or lymphatics. Because of the chronic local destruction of facial structures, MCL may deface the patient if not recognized and adequately treated [2,3]. Patient and observation     A 66-year-old German woman presented in a university hospital with nasal obstruction. Clinically the patient showed nodular and ulcerative lesion at the nasal septum. Histology of the left nasal septum biopsy revealed necrosis, chronic inflammation and intracellular amastigote that was suggestive of leishmaniasis (Figure 1). The molecular-pathological detection of parasite DNA was positive for leishmania infantum. The medical history includes hypothyroidism and rheumatoid arthritis for which she was under continuous immunosuppressive treatment with methotrexate and anti-TNF-blocker (Humira®) several years ago. Moreover, she was treated for cutaneous leishmaniasis in the left elbow after a travel history in Mallorca five years ago (4 weeks, amphotericin B). The patient successfully retreated as a case of mucocutaneous leishmaniasis receiving at first liposomal amphotericin B for 2 weeks. Under the treatment showed the patient an increased level of creatinine, therefore, the therapy was changed to a phospholipid (Hexadecylphosphocholin = Miltefosin®) for 4 weeks with a side effect of vomiting. Follow up visits showed significant improvement with no recurrence. Discussion     Mucocutaneous leishmaniasis is rarely diagnosed in Germany. The majority of the cases are seen in patients living in or visiting endemic regions [3]. The reviewed medical literature through the electronic database Pubmed showed that leishmaniasis was not notifiable in Germany until 2000. Within 2 years 70 cases of leishmaniasis (43 cutaneous or mucocutaneous/mucosal, 27 visceral) were reported. The data were available for 58 patients (35 cutaneous or mucocutaneous/mucosal; 23 visceral). Harms et al. report of 35 patients with MCL or CL, 30 had a travel history to endemic areas and seventeen lesions were located in the face, including mouth and nose [4]. MCL developed as a consequence of cutaneous leishmaniasis. The literature review showed that the favorite sites for manifestations of this disease in the nose were the cartilaginous part of the nasal septum, the anterior portions of nasal fossa, the floor of the nose and the lower turbinate [5,6]. Patients with endonasal leishmaniasis complain nasal obstruction, epistaxis or rhinorrhea-impaired breathing as well as septal perforation. Misdiagnosis may lead to an unfavorable outcome [3,5,6]. Histologically, MCL may mimic other granulomatous or neoplastic Article    Abrar Adnan Suqati et al. PAMJ - 36(292). 17 Aug 2020.  -  Page numbers not for citation purposes. 3 diseases and differentiation is only possible by the morphological features of the parasite [6,7]. In majority of studies, the gold standard method of diagnosis was a biopsy with the molecular detection of parasite DNA by polymerase chain reaction (PCR) [2,8,9]. The primary goals of therapy for leishmaniasis is to prevent the mortality and morbidity. The first line of treatment for MCL is the pentavalent antimony compounds, liposomal amphotericin B or standard amphotericin B while miltefosine is used as a second line of treatment [9,10]. Conclusion     MCL can lead to serious sequela, however, early diagnosis can prevent complications. The aim of this study to raise the awareness among the otolaryngologist in European countries of leishmaniasis of the mucous membranes as a differential diagnosis of mucocutaneous lesions especially in patients with travel history to endemic areas. Competing interests     The authors declare no competing interests. Authors' contributions     All the authors took part in the realization and the implementation of this work. All the authors have read and agreed to the final manuscript. Acknowledgements     We acknowledge support from the German Research Foundation (DFG) and the Open Access Publication Funds of Charité-Universitätsmedizin Berlin. Figure     Figure 1: photomicrograph shows intracellular amastigote (hematoxylin-eosin stain) References     1. World Health Organization. Mucocutaneous leishmaniasis-WHO. 2020. 2. Wollina U, Koch A, Guarneri C, Tchernev G, Lotti T. Cutaneous leishmaniasis - a case series from Dresden. Open Access Maced J Med Sci. 2018;6(1): 89-92. PubMed| Google Scholar 3. Marra F, Chiappetta MC, Vincenti V. Ear, nose and throat manifestations of mucocutaneous Leishmaniasis: a literature review. Acta Biomed. 2014;85(1): 3-7. PubMed| Google Scholar 4. Harms G, Schönian G, Feldmeier H. Leishmaniasis in Germany. Emerging Infect Dis. 2003;9(7): 872-875. PubMed| Google Scholar 5. Aliaga L, Cobo F, Mediavilla JD, Bravo J, Osuna A, Amador JM et al. Localized mucosal leishmaniasis due to Leishmania (Leishmania) infantum: clinical and microbiologic findings in 31 patients. Medicine (Baltimore). 2003;82(3): 147-158. PubMed| Google Scholar 6. Virgilio GR, Hale BR. A case of mucocutaneous leishmaniasis. Otolaryngol Head Neck Surg. 2005;132(5): 800-801. PubMed| Google Scholar 7. González K, Diaz R, Ferreira AF, García V, Paz H, Calzada JE et al. Histopathological characteristics of cutaneous lesions caused by leishmania viannia panamensis in Panama. Rev Inst Med Trop Sao Paulo. 2018;60: e8. PubMed| Google Scholar 8. McGwire BS, Satoskar AR. Leishmaniasis: clinical syndromes and treatment. QJM. 2014;107(1): 7-14. PubMed| Google Scholar 9. Elmahallawy EK, Sampedro Martinez A, Rodriguez-Granger J, Hoyos-Mallecot Y, Agil A, Navarro Mari JM et al. Diagnosis of leishmaniasis. J Infect Dev Ctries. 2014;8(8): 961-972. PubMed| Google Scholar 10. Barratt G, Legrand P. Comparison of the efficacy and pharmacology of formulations of amphotericin B used in treatment of leishmaniasis. Curr Opin Infect Dis. 2005;18(6): 527-530. PubMed| Google Scholar Article    Abrar Adnan Suqati et al. PAMJ - 36(292). 17 Aug 2020.  -  Page numbers not for citation purposes. 4  Figure 1: photomicrograph shows intracellular amastigote (hematoxylin-eosin stain)  

Mucocutaneous leishmaniasis: case report and literature review of a rare endonasal infection

Leishmaniasis is a protozoal infection transmitted by a sandfly vector. In Germany, leishmaniasis of the mucous membranes is a rare condition and usually due to extension of local skin disease into the mucosal tissue via direct extension, bloodstream or lymphatics. We report a case of endonasal leishmaniasis in a female German resident who presented in a university hospital with nasal obstruction. Histology of the left nasal septum biopsy was suggestive of leishmaniasis. The molecular detection of DNA was positive for leishmania infantum. The patient was successfully treated as a case of mucocutaneous leishmaniasis receiving liposomal amphotericin follow up visits showed significant improvement with no recurrence

Mucocutaneous leishmaniasis: case report and literature review of a rare endonasal infection

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