Atypical Lesions as a Sign of Cutaneous Dissemination of Visceral Leishmaniasis in a Human Immunodeficiency Virus–Positive Patient Simultaneously Infected by Two Viscerotropic Leishmania Species

Abstract

Leishmaniasis is considered an emerging opportunistic disease in human immunodeficiency virus (HIV)–infected patients who have considerably variable clinical presentation. We report a patient with visceral leishmaniasis who had unexpected clinical aspects (atypical cutaneous lesions appearing after long-term evidence of visceral parasites). The patient had hepatoesplenomegaly in the absence of fever, but was otherwise generally healthy. The HIV viral load was low despite severe immunossupression (low lymphocyte proliferation and low level of interferon-γ, concomitant with a high lymphocyte activation status). Surprisingly, two Leishmania strains were isolated from his bone marrow (typical L. infantum sequence MON-1, type A) and skin (L. donovani MON-2 sequence); this second strain had not been previously identified in Brazil. The association of visceral leishmaniasis and HIV/acquired immunodeficiency syndrome is a largely unknown disease, particularly in areas in which leishmaniasis is not endemic. Such atypical cases indicate that this disease can be undiagnosed in clinical settings.

Immune Activation and Bacterial Translocation: A Link between Impaired Immune Recovery and Frequent Visceral Leishmaniasis Relapses in HIV-Infected Patients

“…Our group has previously reported that diminished levels of cellular activation were stably maintained in a co-infected patient during the remission phase over a 12-month prospective follow-up period [20]. However, reactivation episodes were again marked by increased activation along with progressively lower levels of specific IFN- γ in response to parasite antigens [20]. Herein, a positive correlation between the number of relapses and the degree of cellular activation was observed in co-infected patients (p<0.05 and r = 0.80, data not shown).…”

“…Considering that such pathogenic features are similar between VL and HIV, these results reinforce the concept that VL may contribute to worsening the immunosuppression induced by HIV infection, accelerating progression to AIDS. In addition, the quality of the Leishmania -specific immune response may remain impaired in Leishmania /HIV patients receiving HAART due to reduced T cell proliferative capacity and deficient interferon (IFN)-γ production [19,20]. Consequently, this deficient parasite control may favor the spread of L .…”

“…infantum to unexpected sites and the appearance of atypical clinical features. In this scenario, amastigotes have been recovered from skin [20,21] and from gut-associated lymphoid tissue (GALT) [22,23]. …”

Determinants for the Development of Visceral Leishmaniasis Disease

“…All of these are associated with impaired immune responses against the parasite. Visceral leishmaniasis in HIV-coinfected individuals can be caused by strains and species that normally cause cutaneous disease [53] and is associated with atypical disease manifestations, such as the presence of cutaneous lesions as well as visceral parasitemia [54].…”

Para-kala-azar dermal leishmaniasis in a patient in Brazil: a case report

“…In recent years, the disease has spread to all regions and emerged in urban areas 2 . In Latin America, cutaneous manifestations in leishmaniasis are related to Leishmania from the subgenus Viannia 3 ; however, there are reports of cutaneous lesions similar to those observed in post-kala-azar dermal leishmaniasis (PKDL), caused by Leishmania infantum, mainly in human immunodeficiency virus (HIV)-infected patients 4,5 . In addition, L. infantum can cause atypical cutaneous leishmaniasis in Central America unrelated to VL 6 .…”