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Indian Journal of Dermatology, Venereology and Leprology
Medknow Publications on behalf of The Indian Association of Dermatologists, Venereologists and Leprologists (IADVL)
ISSN: 0378-6323 EISSN: 0973-3922
Vol. 77, Num. 2, 2011, pp. 251-251

Indian Journal of Dermatology, Venereology, and Leprology, Vol. 77, No. 2, March-April, 2011, pp. 251

Net Case

Unresponsive cutaneous leishmaniasis and HIV co-infection: Report of three cases

Prasoon Soni¹, Neha Prasad¹, Kanika Khandelwal¹, Bhikam C Ghiya¹, Rajesh D Mehta¹, Ram A Bumb¹, Poonam Salotra²

¹ Department of Dermatology, SP Medical College and PBM Group of Hospitals, Bikaner, Rajasthan, India
² Department of Dermatology, Institute of Pathology (ICMR), Safdarjung Hospital, New Delhi, India

Correspondence Address: Ram A Bumb, H-3, PBM Hospital Campus, Bikaner, Rajasthan, India,dr_bumb@rediffmail.com

Code Number: dv11073

PMID: 21393971

DOI: 10.4103/0378-6323.77484

Abstract

Cutaneous leishmaniasis (CL) is a vector borne disease caused by various species of Leishmania parasite. CL is endemic in the Thar desert of Rajasthan state and Himachal Pradesh in India. Immune suppression caused by human immunodeficiency virus (HIV) infection is associated with atypical clinical presentation of CL which responds poorly to the standard treatment and causes frequent relapses. We are reporting three cases of localized and disseminated CL due to Leishmania tropica which failed to respond to conventional intralesional/intramuscular sodium stibogluconate (SSG) injections. Initially, we did not think of HIV infection because CL is endemic in this region. When patients did not respond to SSG injections, we performed enzyme-linked immunosorbent assay (ELISA) tests for HIV and they turned out to be HIV positive. Our report showed that CL is emerging as an opportunistic infection associated with HIV/AIDS and may be the first manifestation in HIV positive patients in an endemic area.

Keywords: Human immunodeficiency virus infection, sodium stibogluconate, unresponsive cutaneous leishmaniasis

Introduction

Cutaneous leishmaniasis (CL) is a protozoan disease caused by various species of Leishmania and transmitted by Phlebotomus sandfly in Old World. The Thar desert of Rajasthan state, Bikaner, located in the northwestern part of India, is one of the endemic areas for CL, where it is caused by Leishmania tropica.[1] In 2005, Sharma et al. ^[2] also reported a new endemic zone of CL from sub-alpine valley along Satluj River in Kinnaur district of Himachal Pradesh. CL manifests as single or multiple non-itchy, painless papules, nodules or plaques, with or without ulceration, usually over exposed areas of body. Once an individual is infected with CL, it is unlikely that he or she will be re-infected by the same species of Leishmania due to development of lifelong immunity. ^[3]

Immune suppression caused by human immunodeficiency virus (HIV) infection is usually associated with multiple disseminated and atypical cutaneous lesions which respond poorly to the standard treatment and causes frequent relapses. ^[4],[5] Various studies have reported association of CL and HIV/AIDS from different countries in the last two decades. ^[6],[7],[8] Chaudhary et al,[9] and Mehta et al,[10] published single case reports of co-infection from India. The recommended treatment of CL is intralesional or systemic antimonials in immuno-competent patients. ^[3] HIV co-infected patients are also treated successfully with antimonials ^[6],[8] and resistance is reported rarely. ^[11] We are reporting three cases of CL who did not respond to sodium stibogluconate (SSG) injections and were later found to be suffering from HIV infection.

Case Reports

Clinical details, investigations done and treatment given before and after detection of HIV infection in all three cases are described in [Table - 1] and [Table - 2].

Case 1

A 40-year-old male farmer presented with numerous painless, non-itchy, discrete, non-tender nodules and plaques of variable size, distributed asymmetrically over limbs and face for last 1 year [Figure - 1] and [Figure - 2]. Past, personal and family history was insignificant. General and systemic examination did not reveal any abnormality. Because of endemicity in this region, we provisionally diagnosed him as a case of disseminated CL, which was confirmed by skin smear and biopsy. Skin biopsy showed mononuclear infiltrate and macrophages laden with LD bodies [Figure - 3]. Species characterization was done by restriction fragment length polymerization (RFLP) and kinetoplast DNA (kDNA) polymerase chain reaction (PCR) tests, which established L. tropica as the causative species. Routine hemogram, renal and hepatic functions, serum amylase and serum venereal disease research laboratory (VDRL) test were normal. Patient was treated with intramuscular injection of SSG but there was no improvement even after 21 injections and new lesions kept on appearing. Because of unresponsiveness, HIV infection was suspected and confirmed by different enzyme-linked immunosorbent assay (ELISA) methods. On close inquiry, the patient revealed history of extramarital sexual contact 5 years back. HAART (zidovudine, lamivudine, and nevirapine) was started along with second course of intramuscular injection of SSG in the same dosage. After 8 days, the patient developed severe pain abdomen radiating to back with vomiting and fever. Serum amylase was raised from 45 units/L to 650 units/L; the patient was diagnosed as a case of pancreatitis which was confirmed by computerized tomography (CT) scan. Injection SSG was stopped but the patient died in the next few days.

Case 2

A 34-year-old male laborer presented with well-defined, non-tender lesions of different size for the last 6 months [Figure - 4]. Clinically, the patient was diagnosed as CL which was confirmed by skin smear, biopsy and species identification by PCR test. He was treated with twice weekly intralesional injection of SSG. Lesions did not improve clinically even after 5 weeks and two more lesions appeared over left leg within 3 months [Inset of [Figure - 4]]. Observing unresponsiveness to treatment and development of new lesions, ELISA test for HIV was performed and found to be positive. Later, he gave history of blood transfusion for some kind of abdominal surgery for abdominal pain 3 years ago. After all the investigations, oral Rifampicin was started at a dose of 1200 mg/day for 6 weeks. HAART was not started because CD4+ count was more than 350 cells/mm ³ . Lesions healed completely in 12 weeks, after which the patient did not return for further follow-up.

Case 3

A 28-year-old laborer presented with single asymptomatic, non-tender lesion for the last 6 months [Figure - 5]. Diagnosis was confirmed microscopically and the patient was treated with intralesional injections of SSG. Lesion did not improve clinically even after 6 weeks, rather three new lesions [Inset of [Figure - 5]] developed within a period of 2 months. Tests for HIV were done which came out to be reactive. Patient refused for viral load, CD4+ count and HAART. After all routine investigations, he was treated with oral Rifampicin 1200 mg/day for 6 weeks. Patient did not respond to treatment and refused for further management.

Discussion

In the Old World, CL is caused by Leishmania. tropica, Leishmania major, Leishmania infantum and Leishmania aethiopica. ^[3] Multiple lesions scattered over different anatomical areas can occur due to multiple bites of sandfly in an immuno-competent person but usually occurs secondary to an underlying deficiency in cellular immunity. In the past, several authors have reported CL in known cases of HIV/AIDS. ^[5],[7],[8] Chaudhary et al,[9] simultaneously diagnosed a case of co-infection. Our cases were having typical papulo-nodulo-ulcerative lesions of CL without any systemic symptoms; we diagnosed them as cases of localized or disseminated CL which is endemic in this region. When they did not respond to treatment satisfactorily and even new lesions appeared, we thought of HIV infection and found them to be positive. CL usually responds well to systemic or intralesional antimonials and relapses are not frequent in immuno-competent patients. ^[3] In co-infected patients also, antimonial treatment is effective and resistance is very rarely reported. ^[11] Niamba et al,[4] and Rosatelli et al,[8] reported successful treatment with antimonials in co-infected patients of HIV with L. infantum and Leishmania braziliensis, respectively. Unresponsiveness to SSG in our cases may be due to difference in species of Leishmania or host immunological factors. The co-infection causes complex immunological disturbances in the patients, ^[11] both by HIV and Leishmania, which may be the reason of unresponsiveness in the cases presented. Our report indicates that CL may be the first manifestation of HIV infection, particularly in endemic areas.

References

1.	Kumar R, Bumb RA, Ansari NA, Mehta RD, Salotra P. Cutaneous leishmaniasis caused by Leishmania tropica in Bikaner, India: Parasite identification and characterization using molecular and immunologic tools. Am J Trop Med Hyg 2007;76:896-901. Back to cited text no. 1 [PUBMED] [FULLTEXT]
2.	Sharma NL, Mahajan VK, Kanga A, Sood A, Katoch VM, Mauricio I, et al. Localized cutaneous leishmaniasis du 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;72:819-24. Back to cited text no. 2 [PUBMED] [FULLTEXT]
3.	Vega-Lopez F, Hay RJ. Parasitic worms and Protozoa. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's Textbook of Dermatology. 8 ^th ed. West Sussex: Wiley-Blackwell; 2010. p. 37.32-43. Back to cited text no. 3
4.	Niamba P, Traore A, Goumbri-Lompo O, Labreze C, Traore-Barro F, Bonkoungou M, et al. Cutaneous Leishmaniasis in HIV patients in Ouagadougou: Clinical and therapeutic aspects. Ann Dermatol Venereol 2006;133:537-42. Back to cited text no. 4
5.	Agostoni C, Dorigoni N, Malfitano A, Caggese L, Marchetti G, Corona S, et al. Mediterranean leishmaniasis in HIV infected patients: Epidemiological, clinical and diagnostic features of 22 cases. Infection 1998;26:93-9. Back to cited text no. 5 [PUBMED]
6.	Alvar J, Canavate C, Gutierrez-Solar B, Jimenez M, Laguna F, Lopez-Velez R, et al. Leishmania and human immunodeficiency virus co-infection: The first 10 years. Clin Microbiol Rev 1997;10:298-319. Back to cited text no. 6
7.	Puig L, Pradinaud R. Leishmania and HIV co-infection: Dermatological manifestations. Ann Trop Med Parasitol 2003;97:107-14. Back to cited text no. 7 [PUBMED]
8.	Rosatelli JB, Souza CS, Soares FA, Foss NT, Roselino AM. Generalized cutaneous leishmaniasis in acquired immunodeficiency syndrome. J Eur Acad Dermatol Venerol 1998;10:229-32. Back to cited text no. 8
9.	Chaudhary RG, Bilimoria FE, Katare SK. Diffuse cutaneous Leishmaniasis: Co-infection with human immuno deficiency virus (HIV). Indian J Dermatol Venereol Leprol 2008;74:641-3. Back to cited text no. 9 [PUBMED]
10.	Mehta V, Balachandran C, Rao R, Dil SK, Indusri L. Diffuse cutaneous Leishmaniasis in HIV. Dermatol Online J 2009;15:9. Available from: http://dermatology.cdlib.org. [Last accessed on 2009 Apr 15]. Back to cited text no. 10
11.	Croft SL, Shyam Sundar, Fairlamb AH. Drug resistance in leishmaniasis. Clin Microbiol Rev 2006;19:111-26. Back to cited text no. 11