Case report | Volume 36, Article 292, 17 Aug 2020 | 10.11604/pamj.2020.36.292.24543

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

Received: 24 Jun 2020 - Accepted: 30 Jun 2020 - Published: 17 Aug 2020

Domain: Otolaryngology (ENT)

Keywords: Cutaneous infection, leishmaniasis, traveler´s diseases

©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. [doi: 10.11604/pamj.2020.36.292.24543]

Available online at: https://www.panafrican-med-journal.com/content/article/36/292/full

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Case report

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

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

 

 

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 parasite 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    Down

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 Up    Down

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 Up    Down

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 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 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 Up    Down

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 Up    Down

The authors declare no competing interests.

 

 

Authors' contributions Up    Down

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 Up    Down

We acknowledge support from the German Research Foundation (DFG) and the Open Access Publication Funds of Charité-Universitätsmedizin Berlin.

 

 

Figure Up    Down

Figure 1: photomicrograph shows intracellular amastigote (hematoxylin-eosin stain)

 

 

References Up    Down

  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

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

Case report

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

Case report

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

Case report

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

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Key words

Cutaneous infection

Leishmaniasis

Traveler´s diseases

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