Correspondence

Indian Mucosal Leishmaniasis Due to Leishmania donovani Infection

N Engl J Med 2008; 358:313-315January 17, 2008

Article

To the Editor:

A 62-year-old man from Chamoli District of Uttaranchal, north India, had a 2-year history of a fleshy, polypoid swelling with superficial ulceration on the mucosal aspect of the lower lip, lower gum, floor of the mouth, and palate, without involvement of the skin (Figure 1A and 1BFigure 1Lesions before and after Treatment in a Patient with Mucosal Leishmaniasis and Results of a Polymerase-Chain-Reaction (PCR) Assay of a Skin-Lesion Specimen.). He did not have a history of kala-azar. Repeated biopsy of the lip and alveolar mucosa showed an infiltrate composed of lymphocytes, histiocytes, and many plasma cells. Staining for kappa and lambda light chains revealed polyclonal plasma cells. No M band was found on serum immunoelectrophoresis, but there was polyclonal hypergammaglobulinemia. Urinalysis was negative for Bence Jones proteins. Contrast-enhanced computed tomography revealed mild mucosal thickening in the right maxillary sinus, without bony destruction. A smear from a lesion showed structures resembling the amastigote form of Leishmania donovani. A strip test based on the recombinant K39 antigen was strongly positive. A fragment of L. donovani–specific kinetoplast mini-circle DNA, 600 bp in length, was amplified from the skin sample by the polymerase-chain-reaction assay (Figure 1C), as previously described.1 Treatment with sodium stibogluconate (20 mg per kilogram of body weight per day) was started; the lesion improved within a few days, remarkably so within 2 weeks (Figure 1B).

L. donovani is known to cause kala-azar. In 10 to 20% of infections, there is reversal of the parasites from the viscerotrophic to the dermatotrophic form, which results in post–kala-azar dermal leishmaniasis.1-3 Localized cutaneous leishmaniasis due to L. donovani infection has been reported in Kenya, Iraq, and Sri Lanka.4 Recently, a large number of cases of localized cutaneous leishmaniasis due to L. donovani infection were reported in Himachal Pradesh, North India.4 Primary mucosal leishmaniasis has been described in Sudan5 but is exceedingly rare in India.

Our patient had only mucosal involvement due to L. donovani infection. The excellent response to treatment within a few days and nearly complete remission within 2 weeks suggest that the lesion was primary mucosal leishmaniasis rather than the unusual variant of mucosal post–kala-azar dermal leishmaniasis, in which the lesions take some months to subside. The strongly positive result of serologic testing with the recombinant K39 antigen in this patient is characteristic of L. donovani infections in patients with kala-azar or post–kala-azar dermal leishmaniasis. It is possible that some of the Indian strains within the L. donovani complex possess the characteristic genomic sequence associated with primary mucosal leishmaniasis, as described in a recent report from Sudan.5

Gomathy Sethuraman, M.D.
Vinod K. Sharma, M.D.
All India Institute of Medical Sciences, New Delhi 110 29, India
kgsethu@yahoo.com

Poonam Salotra, Ph.D.
Institute of Pathology, New Delhi 110 29, India

5 References

1. 1

   Salotra P, Sreenivas G, Pogue GP, et al. Development of a species-specific PCR assay for detection of Leishmania donovani in clinical samples from patients with kala-azar and post-kala-azar dermal leishmaniasis. J Clin Microbiol 2001;39:849-854
   CrossRef | Web of Science | Medline

2. 2

   Ansari NA, Ramesh V, Salotra P. Interferon (IFN)-gamma, tumor necrosis factor-alpha, interleukin-6, and IFN-gamma receptor 1 are the major immunological determinants associated with post-kala azar dermal leishmaniasis. J Infect Dis 2006;194:958-965
   CrossRef | Web of Science | Medline

3. 3

   Salotra P, Singh R. Challenges in the diagnosis of post kala-azar dermal leishmaniasis. Indian J Med Res 2006;123:295-310
   Web of Science | Medline

4. 4

   Sharma NL, Mahajan VK, Kanga A, et al. Localized cutaneous leishmaniasis due to Leishmania donovani and Leishmania tropica: preliminary findings of the study of 161 new cases from a new endemic focus in Himachal Pradesh, India. Am J Trop Med Hyg 2005;72:819-824
   Web of Science | Medline

5. 5

   Mahdi M, Elamin EM, Melville SE, et al. Sudanese mucosal leishmaniasis: isolation of a parasite within the Leishmania donovani complex that differs genotypically from L. donovani causing classical visceral leishmaniasis. Infect Genet Evol 2005;5:29-33
   CrossRef | Medline

Citing Articles (2)

Citing Articles

1. 1

   Deepani Rathnayake, Ranthilaka R. Ranawake, Ganga Sirimanna, Yamuna Siriwardhane, Nadeera Karunaweera, Rajiv De Silva. (2010) Co-infection of mucosal leishmaniasis and extra pulmonary tuberculosis in a patient with inherent immune deficiency. International Journal of Dermatology 49:5, 549-551
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2. 2

   Angamuthu Selvapandiyan, Robert Duncan, Juan Mendez, Rajesh Kumar, Poonam Salotra, Lisa J. Cardo, Hira L. Nakhasi. (2008) A Leishmania minicircle DNA footprint assay for sensitive detection and rapid speciation of clinical isolates. Transfusion 48:9, 1787-1798
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