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CASE REPORT
Year : 2006  |  Volume : 51  |  Issue : 4  |  Page : 278-280
Borderline tuberculoid leprosy: A manifestation of immune reconstitution inflammatory syndrome in a human immunodeficiency virus infected person


1 Consultant Dermatologist, Chandannagar, Hooghly, West Bengal, India
2 Department of Tropical Medicine, School of Tropical Medicine, Kolkata - 700037, India
3 Department of Dermatology, Midnapur Medical College, Midnapur, West Bengal, India

Correspondence Address:
Partha Mukhopadhyay
Purano Bajir Karkhana, Dupliex Patty Main Road, Chandannagar, Hooghly, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.30295

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   Abstract 

Immune reconstitution inflammatory syndrome describes a collection of inflammatory disorders associated with paradoxical deterioration of various pre-existing processes following start of highly active antiretroviral therapy (HAART) in human immunodeficiency virus (HIV)-infected patients. Leprosy as an opportunistic infection in immune reconstitution syndrome has been rarely reported in literature. A case of a 30-year-old HIV positive man with extrapulmonary tuberculosis of left foot on HAART having developed borderline tuberculoid leprosy as opportunistic infection in immune reconstitution syndrome has been reported.


Keywords: Highly active antiretroviral therapy, immune reconstitution inflammatory syndrome, leprosy


How to cite this article:
Mukhopadhyay P, Pal S, Mallik S, Biswas S, Saha B. Borderline tuberculoid leprosy: A manifestation of immune reconstitution inflammatory syndrome in a human immunodeficiency virus infected person. Indian J Dermatol 2006;51:278-80

How to cite this URL:
Mukhopadhyay P, Pal S, Mallik S, Biswas S, Saha B. Borderline tuberculoid leprosy: A manifestation of immune reconstitution inflammatory syndrome in a human immunodeficiency virus infected person. Indian J Dermatol [serial online] 2006 [cited 2019 Nov 22];51:278-80. Available from: http://www.e-ijd.org/text.asp?2006/51/4/278/30295



   Introduction Top


Immune reconstitution inflammatory syndrome (IRIS) describes a collection of different inflammatory disorders which are associated with paradoxical deterioration of various pre-existing infectious processes following commencement of highly active antiretroviral therapy (HAART) in HIV-infected patients.[1]

Various opportunistic infections may be a presentation of immune reconstitution in these patients. Very few cases of leprosy as the opportunistic infection in immune reconstitution syndrome has been reported in literature.[2],[4],[5],[6] Here we are reporting a case of boderline tuberculoid leprosy with type 1 reaction developed in a HIV-infected patient with extrapulmonary tuberculosis of left foot 2.5 months after starting highly active antiretroviral l therapy.


   Case Report Top


A 30-year-old unmarried man, owner of a small grocery shop, had initially presented to his general physician with low grade fever along with swelling of left foot for about one year. Routine conservative treatment done by the physician showed no improvement. X-ray left foot showed an erosion of the second metatarsal and proximal phalanx. He was referred to an orthopaedician. Biopsy done by the orthopaedician showed tuberculous granuloma. The patient was put on AT drugs with rifampicin, INH, ethambutol and pyrazinamide.

A history of high-risk behaviour prompted an HIV test being done and it was reactive. The patient was referred to us at this stage.

On examination the patient was mildly anaemic, with no significant lymphadenopathy, no genital ulcer. Oral candidiasis was present. Respiratory, cardiovascular, gastrointestinal and neurological examination was normal. The left forefoot showed a local swelling with a small ulcer 1cm by 0.5 cm at biopsy site with serosanguinous discharge.

Investigations revealed the following- Routine hemogram-Hb-11 gram%, Total leukocyte count-5320/cu mm. with poly-74%, lympho-22%, monocyte-2%, eosinophil-2%. ESR-86 mm/1sth (Westergren). Fasting plasma glucose-89 mg%, creatinine-0.8 mg%, serum albumin-3.4 mg%, globulin-4.8 mg%, SGPT-38 IU/l, alkaline phosphatase-186 IU/l (normal <171 IU/l).

HIV serology- reactive by two different ELISAs, confirmed by Western Blot. CD4 count-111 cells/ml, VDRL-Non reactive, HbsAg-negative, anti HCV-negative. Viral load could not be done due to financial constraints.

X-ray left foot -Erosion of the second metatarsal and proximal phalanx.

Review of Biopsy slide of left foot swelling- Tuberculous granuloma.

Culture from serosanguinous discharge from left foot ulcer- No growth. Sputum for AFB -negative. Mantoux test with 5 TU PPD was negative.

Chest X-ray- Hilar lymphadenopathy.

The patient was already on AT drugs for two weeks. He tolerated drugs well. Fluconazole 100 mg daily was given for 10 days. He was put on cotrimoxazole prophylaxis. Appropriate HAART regimen consisting of lamivudine, stavudine and efavirenz was started. After two months of 4-drug ATD, ethambutol and pyrazinamide were discontinued and he was put on continuation phase with rifampicin and INH.

After 2.5 months of treatment with ART, the patient developed erythematous infiltrative skin lesions on right forearm and back. His local doctor initially treated him as drug allergy and subsequently referred to us. He had two anaesthetic plaques without any nerve thickening. Skin smear was negative for acid fast bacilli [Figure - 1].

Skin biopsy from the back lesion revealed typical histological features of BT leprosy [Figure - 2]. His CD 4 count at this time was 185 cells/ml. Since he was getting daily rifampicin for tuberculosis, dapsone was added to the treatment regime. After completion of antitubercular therapy monthly rifampicin was added. Absence of nerve involvement prompted us to use only routine non-steroidal anti inflammatory drugs as anti inflammatory treatment.

Antileprotic drugs were stopped after six months. Regular follow-up showed subsidence of leprosy lesions. He is presently doing well and last CD4 count on 9.6.05 was 276 cells/ml.


   Discussion Top


IRIS is an unusual inflammatory reaction to an opportunistic infection that occurs in HIV positive patients with profound immunosuppression during the reconstitution of the immune system in the initial months of highly active antiretroviral treatment.[2]

Little is known about the specific immune mechanisms involved in the pathogenesis of immune reconstitution disorders (IRD) and these mechanisms vary from one opportunistic infection to another. Nevertheless, many immune reconstitution syndromes appear to result from enhanced antigen-specific immune responses that lead to increased production of inflammatory mediators.[3]

Tuberculosis IRD is the most commonly occurring IRD syndrome worldwide.[3] Among other immune reconstitution disorders, MAC, CMV, cryptococcal IRD, herpes simplex, herpes zoster have been encountered.[3],[4] Regarding leprosy, only a handful of cases as a manifestation of IRIS have been published.

Three cases of leprosy with reactional states have been reported in one series.[2] Three cases of atypical leprosy has also been reported amongst dermatological manifestations associated with IRIS.[4] One solitary case each of tuberculoid leprosy have been reported by two different set of authors.[5],[6]

The association between development of the borderline tuberculoid lesions with type-1 reaction and changes in blood CD4+ lymphocyte count observed during HAART in our case strongly suggests that the presentation of leprosy in reaction resulted from immune reconstitution. The disease might have been present previously and following immune recovery with HAART a type-1 reaction occurred in the lesions making it visible to the patient. The patient was receiving rifampicin as part of antitubercular regimen, which might have possibly precipitated a type-1 reaction.

It is important to note that most IRD cases have resolved within several weeks simply by continuing ART and treatment of opportunistic pathogens.[3] In our case also, continuation of HAART along with treatment for leprosy resulted in subsidence of leprosy lesions.

 
   References Top

1.Goebel FD. Immune reconstitution inflammatory syndrome (IRIS): Another new disease entity following treatment initiation of HIV infection. Infection 2005;33:43-5.  Back to cited text no. 1      
2.Couppie P, Abel S, Voinchet H, Roussel M, Helenon R, Huerre M, et al . Immune reconstitution inflammatory syndrome associated with HIV and leprosy. Arch Dermatol 2004;140:997-1000.  Back to cited text no. 2      
3.Jacobson MA. University of California San Francisco Available from: http://hivinsite.ucsf.edu/insite?page=kb03-04-03 HIV In Site Knowledge Base Chapter. 2005.  Back to cited text no. 3      
4.Sarazin E, Nacher M, Toure Y, Clyti E, El Guedj M, Aznar C, et al . Dermatologic manifestation associated with immune reconstitution syndrome in HIV+ patients starting HAART: A retrospective study in French Guiana. Bull Soc Pathol Exot 2005;98:187-92.  Back to cited text no. 4      
5.Visco-Comandini U, Longo B, Cuzzi T, Paglia MG, Antonucci G. Tuberculoid leprosy in a patient with AIDS: A manifestation of immune restoration syndrome. Scand J Infect Dis 2004;36:881-3.  Back to cited text no. 5      
6.Lawn SD, Wood C, Lockwood DN. Borderline tuberculoid leprosy: An immune reconstitution phenomenon in a human immunodeficiency virus-infected person. Clin Infect Dis 2003;36:e5-6.  Back to cited text no. 6      


    Figures

  [Figure - 1], [Figure - 2]

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    Abstract
    Introduction
    Case Report
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    References
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