Isolated mucosal Leishmaniasis

Abstract

Leishmaniasis is a term used to define a group of clinical syndrome caused by various species of parasite Leishmania. Three main clinical types of leishmaniasis are visceral leishmaniasis, cutaneous leishmaniasis and mucocutaneous leishmaniasis. However, isolated presentation as mucosal disease is rare. We report a case of primarily mucosal leishmaniasis.

Keywords: Leishmania donovani, mucosal leishmaniasis, rK-39 antibody

How to cite this article:
Sundriyal D, Kumar N, Kumar R, Sharma B. Isolated mucosal Leishmaniasis. Ann Trop Med Public Health 2013;6:487-8

 

How to cite this URL:
Sundriyal D, Kumar N, Kumar R, Sharma B. Isolated mucosal Leishmaniasis. Ann Trop Med Public Health [serial online] 2013 [cited 2020 Oct 31];6:487-8. Available from: https://www.atmph.org/text.asp?2013/6/4/487/127808

 

Introduction

Leishmaniasis is endemic in India. Visceral leishmaniasis is the major form in which the disease occurs and is caused by Leishmania donovani species usually in immunosuppressed patients. Mucosal variety of the disease is rare in this part of the world. Mucosal leishmaniaisis can be the initial presenting feature in an immunosuppressed patient. We present a case of isolated mucosal leishmaniasis in an immunocompetent host who was treated successfully.

Case Report

A 42-year-old male, resident of Uttar Pradesh, India and a laborer by occupation presented with history of gradually increasing difficulty in swallowing for 6 months which was associated with hoarseness of voice for the last 3 months. He was a chronic smoker for the last 15 years. There was no history of fever, weight loss, or hemoptysis. Past and family histories for any chronic illness or treatment undergone were unremarkable. On examination, the patient was conscious, oriented and pale. No palpable lymph nodes or skin lesions were seen. On examination of the oral cavity, there was a growth involving the hard palate, soft palate, and uvula [Figure 1]. Tonsils were enlarged. Examination of the chest, the abdomen and the cardiovascular system was normal.

Figure 1: Mucosal leishmaniasis caused by Leishmania donovani. Initial presentation of the growth of hard and soft palates

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Investigations revealed hemoglobin of 11-g/dL, total leucocyte count of 4100/cumm, differential count P/L/E/M 62/34/4/0, Erythrocyte Sedimentation Rate: 53mm 1 st h. Liver and kidney function tests were normal. ELISA for HIV 1 and 2 were non-reactive. Skiagram of chest was normal. Ultrasonography of the abdomen was also normal. A palatal biopsy was taken which was suggestive of granulomatous reaction with abundant eosinophilia, lymphocytosis and plasma cells. There was no evidence of malignancy. Smears were negative for acid-fast-bacilli. On Geimsa staining, organisms morphologically resembling Leishmania donovani were seen in abundance.

rK39 antibody test was positive. Bone marrow aspiration smears were negative for Leishmania donovani. Patient was diagnosed as a case of isolated mucosal leishmaniasis and was treated with amphotericin B in the dose of 0.7-mg/kg/day for 20 days. His palatal growth regressed and hoarseness disappeared in due course of treatment [Figure 2].

Figure 2: Follow-up presentation after treatment showing regression of the palatal growth

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Discussion

The primarily mucosal variety of leishmaniasis is a very uncommon illness and only a few cases have been reported. [1],[2] Leishmaniasis has been seen usually in immunosuppressed patients due to various causes like HIV infection, diabetes, neoplasms, and so on. Garcia de Marcos and Ferrer reported a series of localized leishmaniasis of oral mucosa in immunosuppressed patients due to HIV and diabetes. [3] In Indian subcontinent, visceral form of the disease is seen and only a few cases of primary mucosal leishmaniasis due to Leishmania donovani have been reported. [4],[5] Post -kala-azar dermal leishmaniasis can present as cutaneous mucosal lesions, but the clinical features and long duration of treatment differentiate it from primary mucosal disease. It is worth noting that primary mucosal variety of leishmaniasis is usually seen in South American countries and is caused by Leishmania braziliensis, whereas in Indian subcontinent it is very rare and is caused by Leishmania donovani. It is, thus, obvious that organism commonly associated with visceral disease can cause primary mucosal disease as well. It is of clinical interest that a possibility of leishmaniaisis should be kept in mind in patients presenting with mucosal lesions specially those with immunosuppressed status in our country or in those areas where the parasite is endemic. A complete examination of the upper respiratory tract of patients with mucosal leishmaniasis is necessary to establish the extent and the severity of the disease. Our patient was, however, immunocompetent and still developed mucosal disease.

References

 

1. Leitner V, Weingast J, Harmankaya K, Walochnik J, Pehamberger H, Petzelbauer P, et al. Leishmaniasis in the tongue of an immunocompetent man. Am J Trop Med Hyg 2010;82:597-9.
2. Osorio LE, Castillo CM, Ochoa MT. Mucosal leishmaniasis due to Leishmania(Viannia) panamensis in Colombia : Clinical characteristics. Am J Trop Med Hyg 1998;59:49-52.
3. Garcia de Marcos JA, Dean Ferrer A, Alamillos Granados F, Ruiz Masera JJ, Cortes Rodríguez B, Vidal Jimenez A, et al. Localized leishmaniasis of the oral mucosa. A report of three cases. Med Oral Patol Oral Cir Bucal 2007;12:E281-6.
4. Sethuraman G, Sharma VK, Salotra P. Indian mucosal leishmaniasis due to Leishmania donovani infection. N Engl J Med 2008;358:313-5.
5. Kumar B, Ghimire A, Karki S, Upadhyaya P. Primary laryngeal leishmaniaisis: A rare case report. Indian J Pathol Microbiol 2009;52:62-4.

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/1755-6783.127808

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[Figure 1], [Figure 2]

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