Post-kala-azar dermal Leishmaniasis in two different clinical contexts^*

Abstract

In Brazil, visceral Leishmaniasis is caused by Leishmania chagasi. The development of cutaneous lesions in visceral leishmaniasis patients has been described in two different clinical contexts. Patients with compromised immunity can develop skin lesions as a direct consequence of a current visceral disease. Equally, patients with a history of kala-azar and progressive, immune improvement occasionally develop skin lesions as a consequence of immune reconstitution infl ammatory syndrome. These cases manifest in similar fashion to the classic form of post-kala-azar dermal Leishmaniasis. We describe different cases that exemplify these two clinical presentations.

Keywords:
Dermatology; HIV; Immune reconstitution inflammatory syndrome; Leishmania infantum; Leishmaniasis; Skin diseases

INTRODUCTION

The development of skin lesions in the context of Leishmania sp and HIV co-infection has been described in two different situations.^1,2Habitually, cutaneous involvement occurs after starting of highly active antiretroviral therapy (HAART) in the context of immune reconstitution syndrome.¹Dermatological lesions can also occur due to a current kala-azar episode.²

CASE REPORTS

Case 1

A male AIDS sufferer of 6 years, aged 35, needed assistance at the dermatology department during antiretroviral therapy, due to erythematous nodules, some of which were confluent, leading to infiltrated patches with mild scaling that had emerged one year and three months earlier (Figures 1and2). The lesions were disseminated. At that point, he did not present systemic signs or symptoms. He had experienced a visceral Leishmaniasis episode four years previously, treated with Amphotericin B, and subsequently underwent prophylaxis with this drug, due to frequent relapses of the disease. Histopathological examination of the skin sample showed amastigote forms of Leishmania sp. The PCR for Leishmania chagasi, using the Primers RV1 and RV2, was positive and the Montenegro's reaction was 14mm. At the time, the CD4+ cell count was 330cel/mm³, while the CD4+ cell count was 92cels/mm³, three months before lesions developed. This patient was treated using Amphotericin B, with complete resolution of skin lesions.

FIGURE 1
Papular and nodular lesions on the trunk. They were also present on the extremities and back

FIGURE 2
Facial lesions

Case 2

A male, aged 42, HIV-positive for 13 years, undergoing irregular use of antiretroviral therapy, sought assistance due to fever, weight loss and skin lesions that had appeared in the two preceding months. Physical examination revealed disseminated, slightly erythematous papules and nodules, as well as papular lesions on the palate (Figures 3and4). He had a 4-year history of visceral Leishmaniasis and since then, had been under prophylactic therapy with amphotericin B. At that point, the CD4+ cell count was 56 cells/mm³, while two months before skin lesions developed, the CD4+ cell count was 108cells/mm³. Leishmania sp amastigotes were present on the myelogram, while the PCR, using the RV1 and RV2 primers, was positive in peripheral blood. The patient was treated with Lipossomal Amphotericin B, leading to complete resolution of the visceral and cutaneous condition.

FIGURE 3
Papules and nodules on the abdomen. Similar lesions were present throughout the body

FIGURE 4
Lesions on the palate

DISCUSSION

We present two rare cases of disease association. The development of skin lesions in patients with HIV/Kala-azar coinfection has been described in two different situations.^1,2

Cutaneous lesions can develop in the context of immune reconstitution inflammatory syndrome (IRIS), caused by Leishmania.¹IRIS comprises a group of disorders that occur after some degree of immune restoration, which results in pathological inflammation to pathogens and their antigens.³Clinical manifestations depend on the agent involved.^3,4The diagnosis is clinical and opportunistic infections should be considered in the differential.⁴In HIV-positive individuals, one essential criterion for diagnosis is that patients be receiving antiretroviral therapy, with strong virological and immunological responses, meaning an increase in CD4+ cell count from the baseline and a decrease in HIV RNA level.³

IRIS cases related to Leishmania generally present with post-kala-azar dermal Leishmaniasis (PKDL), with a mean CD4⁺ cell count of 175 cells/mm³ when skin lesions emerge.¹Similarly to the classical form of PKDL, the ability to mount an immunologic response to the parasite still present in skin has been implicated in the development of cutaneous lesions.⁵Papules and nodules that are commonly located on the cephalic region typically occur at variable moments once the visceral disease is cured.⁶Case 1 is an example of PKDL in the context of IRIS, as demonstrated by the increasing CD4+ cell counts and the good immunologic response, confirmed by the positive Montenegro’s skin test. Furthermore, the patient did not show signs of visceral involvement, which would be expected in a case of visceral Leishmaniasis with secondary skin involvement.

Another possible dermal presentation of the HIV/kala-azar co-infection is the emergence of disseminated, cutaneous lesions during an active, visceral Leishmaniasis episode¹. The most common symptoms of kala-azar in HIV-positive individuals are paleness (91.4% of cases) and fever (85.9% of cases).⁷Moreover, patients with active infection habitually present moderate to severe immunosuppression and the mean CD4⁺ count is between 44 and 123 cells.^8,9,10

Patient 2 presented signs of visceral involvement caused by Leishmania and his CD4+ cell count was decreasing. This data support the diagnosis of a kala-azar episode with secondary skin involvement in a patient with progressive immune depletion.

• Conflict of Interest: None.
• How to cite this article: Barroso DH, Silva CEF, Vasconcelos ACDP, Cavalcanti SMM, Brito MEF, Medeiros ACR. Post-Kala-azar dermal Leishmaniasis in two different clinical contexts. An Bras Dermatol. 2015;90 (3 Suppl 1):S108-10
• *
  Work performed at the Hospital Universitário Oswaldo Cruz - Universidade de Pernambuco (UPE) - Recife (PE), Brazil.

References

• ¹
  Antinori S, Longhi E, Bestetti G, Piolini R, Acquaviva V, Foschi A, et al. Post-kala-azar dermal leishmaniasis as an immune reconstitution inflammatory syndrome in a patient with acquired immune deficiency syndrome. Br J Dermatol. 2007;157:1032-6.
• ²
  González-Beato MJ, Moyano B, Sánchez C, González-Beato MT, Pérez-Molina JA, Miralles P, et al. Kaposi's sarcoma-like lesions and other nodules as cutaneous involvement in AIDS-related visceral leishmaniasis. Br J Dermatol. 2000;143:1316-8.
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  Alexandrino-de-Oliveira P, Santos-Oliveira JR, Dorval ME, Da-Costa Fd, Pereira GR, da Cunha RV, et al. HIV/AIDS associated visceral leishmaniasis in patients from an endemic area in Centralwest Brazil. Mem Inst Oswaldo Cruz. 2010;105:692-7.
• ¹⁰
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Publication Dates

• Publication in this collection
  June 2015

History

• Received
  15 Jan 2014
• Accepted
  30 Mar 2014