AIDS RESEARCH AND HUMAN RETROVIRUSES Volume 30, Number 9, 2014 ª Mary Ann Liebert, Inc. DOI: 10.1089/aid.2014.0118
Unusual Presentation of Visceral Leishmaniasis in an HIV-Infected Patient Giovanni Cenderello,1 Emanuele Pontali,1 Corrado Ruggeri,2 Andrea Dusi,3 and Andrea De Maria 4,5
FIG. 1.
V
Lymph node. Leishmania organisms are within the cytoplasm of the macrophages. H&E 800 · .
isceral leishmaniasis (VL) is an infectious disease caused by Leishmania infantum and is endemic in 88 countries. It is usually associated with fever, anemia, and splenomegaly, commonly defined to as ‘‘cardinal signs.’’1,2 In areas of relatively low incidence, and with higher economic 1
Department Department 3 Department 4 Department 5 Department 2
of of of of of
development, it can occur more frequently than expected in immune-suppressed subjects, e.g., human immunodeficiency virus (HIV)-infected transplant recipients. In these cases, the clinical presentation, diagnosis, and management may be challenging. In fact, a wide spectrum of clinical presentations
Infectious Diseases, Galliera Hospital, Genoa, Italy. Pathology, ASL 1 Imperiese, Sanremo, Italy. Microbiology, ASL 1 Imperiese, Sanremo, Italy. Infectious Diseases, IRCCS Sann Martino-IST, Genoa, Italy. Health Sciences, University of Genoa, Genoa, Italy.
846
ATYPICAL VISCERAL LEISHMANIASIS
has been recently reported in immunocompromised adults.3 Lymphoid localization of Leishmania is a known, albeit rare, manifestation in immune-competent patients and is classified as ‘‘paucisyntomatic leishmaniasis’’ with no visceral dissemination.4 Figure 1 presents a lymphonodal biopsy showing a lymphoadenopathy sustained by Leishmania. The patient, a 55-year-old man, had HIV and was receiving antiretroviral therapy with emtricitabine, tenofovir, atazanavir, and ritonavir. The treatment had been successful, achieving an undetectable HIV viral load ( < 50 HIV-RNA copies/ml); nevertheless, immunological discordance was observed with a modest immune recovery (CD4 + count always < 300 cells/mm3). The patient was hospitalized because of persistent fever and leucopenia; during hospitalization splenomegaly and a generalized enlargement of the lymph nodes were detected and confirmed at CT scan, leading to a workup to investigate a possible hematological malignancy. A lymph node biopsy showed the presence of Leishmania spp. (Fig. 1). The biopsy showed a pattern characterized by Leishmania amastigotes. In addition, Leishmania antigen was detected in urine samples and Leishmania serology (IFAT) was positive. A bone marrow biopsy revealed intracellular parasites on Giemsa staining. The patient was treated with liposomal amphotericin B and rapidly recovered. Thus, diffuse lymphoadenopathy sustained by Leishmania infantum does not always represent a benign localized disease, and may be associated with a complex clinical presentation as previously reported.5 In immune-suppressed subjects with Leishmania lymphadenopathy, bone marrow and urinary antigen evaluation are indicated. The present case was observed and evaluated within the frame of a clinical regional registry, Registro Ligure Leishmaniosi Viscerale (RiLLeVI). In areas of endemicity with a relatively low incidence, long-term registries collecting viral load cases are a useful tool to maintain a clinical perspective, to share experience concerning unusual manifestations of the disease, and to record specific local epidemiological trends.
847 Author Disclosure Statement
No competing financial interests exist. References
1. Peachey AM, Irvine GH, and White H: Visceral leishmaniasis: An unusual cervical presentation. Br J Oral Maxillofac Surg 1994;32(5):325–327. 2. Dujardin JC, Campino L, Can˜avate C, Dedet JP, Gradoni L, Soteriadou K, Mazeris A, Ozbel Y, and Boelaert M: Spread of vector-borne diseases and neglect of leishmaniasis, Europe. Emerg Infect Dis 2008;14(7):1013–1018. 3. Cenderello G, Pasa A, Dusi A, Dentone C, Toscanini F, Bobbio N, Bondi E, DelBono V, Izzo M, Riccio G, Anselmo M, Giacchino R, Marazzi MG, Pagano G, Cassola G, Viscoli C, Ferrea G, and De Maria A: Varied spectrum of clinical presentation and mortality in a prospective registry of visceral leishmaniasis in a low endemicity area of Northern Italy. BMC Infect Dis 2013;13(1):248. 4. Pomares-Estran C, Cenderello G, Ittel A, Karsenti JM, Cardot-Leccia N, VassaloM, Hasseine L, Delaunay P, Rosenthal E, and Marty P: Isolated lymphadenopathy in Leishmania infantum infection: Three case reports. Ann Trop Med Parasitol 2009;103(6):555–559. 5. Mishra S., Shukla A, Tripathi AK, et al.: Visceral leishmaniasis with HIV coinfection and cervical lymphadenopathy. BMJ Case Rep, published online. Accessed May 7, 2014.
Address correspondence to: Giovanni Cenderello Department of Infectious Diseases Galliera Hospital Via Volta 8 Genoa 16128 Italy E-mail: [email protected]
Unusual Presentation of Visceral Leishmaniasis in an HIV-Infected Patient Giovanni Cenderello,1 Emanuele Pontali,1 Corrado Ruggeri,2 Andrea Dusi,3 and Andrea De Maria 4,5
FIG. 1.
V
Lymph node. Leishmania organisms are within the cytoplasm of the macrophages. H&E 800 · .
isceral leishmaniasis (VL) is an infectious disease caused by Leishmania infantum and is endemic in 88 countries. It is usually associated with fever, anemia, and splenomegaly, commonly defined to as ‘‘cardinal signs.’’1,2 In areas of relatively low incidence, and with higher economic 1
Department Department 3 Department 4 Department 5 Department 2
of of of of of
development, it can occur more frequently than expected in immune-suppressed subjects, e.g., human immunodeficiency virus (HIV)-infected transplant recipients. In these cases, the clinical presentation, diagnosis, and management may be challenging. In fact, a wide spectrum of clinical presentations
Infectious Diseases, Galliera Hospital, Genoa, Italy. Pathology, ASL 1 Imperiese, Sanremo, Italy. Microbiology, ASL 1 Imperiese, Sanremo, Italy. Infectious Diseases, IRCCS Sann Martino-IST, Genoa, Italy. Health Sciences, University of Genoa, Genoa, Italy.
846
ATYPICAL VISCERAL LEISHMANIASIS
has been recently reported in immunocompromised adults.3 Lymphoid localization of Leishmania is a known, albeit rare, manifestation in immune-competent patients and is classified as ‘‘paucisyntomatic leishmaniasis’’ with no visceral dissemination.4 Figure 1 presents a lymphonodal biopsy showing a lymphoadenopathy sustained by Leishmania. The patient, a 55-year-old man, had HIV and was receiving antiretroviral therapy with emtricitabine, tenofovir, atazanavir, and ritonavir. The treatment had been successful, achieving an undetectable HIV viral load ( < 50 HIV-RNA copies/ml); nevertheless, immunological discordance was observed with a modest immune recovery (CD4 + count always < 300 cells/mm3). The patient was hospitalized because of persistent fever and leucopenia; during hospitalization splenomegaly and a generalized enlargement of the lymph nodes were detected and confirmed at CT scan, leading to a workup to investigate a possible hematological malignancy. A lymph node biopsy showed the presence of Leishmania spp. (Fig. 1). The biopsy showed a pattern characterized by Leishmania amastigotes. In addition, Leishmania antigen was detected in urine samples and Leishmania serology (IFAT) was positive. A bone marrow biopsy revealed intracellular parasites on Giemsa staining. The patient was treated with liposomal amphotericin B and rapidly recovered. Thus, diffuse lymphoadenopathy sustained by Leishmania infantum does not always represent a benign localized disease, and may be associated with a complex clinical presentation as previously reported.5 In immune-suppressed subjects with Leishmania lymphadenopathy, bone marrow and urinary antigen evaluation are indicated. The present case was observed and evaluated within the frame of a clinical regional registry, Registro Ligure Leishmaniosi Viscerale (RiLLeVI). In areas of endemicity with a relatively low incidence, long-term registries collecting viral load cases are a useful tool to maintain a clinical perspective, to share experience concerning unusual manifestations of the disease, and to record specific local epidemiological trends.
847 Author Disclosure Statement
No competing financial interests exist. References
1. Peachey AM, Irvine GH, and White H: Visceral leishmaniasis: An unusual cervical presentation. Br J Oral Maxillofac Surg 1994;32(5):325–327. 2. Dujardin JC, Campino L, Can˜avate C, Dedet JP, Gradoni L, Soteriadou K, Mazeris A, Ozbel Y, and Boelaert M: Spread of vector-borne diseases and neglect of leishmaniasis, Europe. Emerg Infect Dis 2008;14(7):1013–1018. 3. Cenderello G, Pasa A, Dusi A, Dentone C, Toscanini F, Bobbio N, Bondi E, DelBono V, Izzo M, Riccio G, Anselmo M, Giacchino R, Marazzi MG, Pagano G, Cassola G, Viscoli C, Ferrea G, and De Maria A: Varied spectrum of clinical presentation and mortality in a prospective registry of visceral leishmaniasis in a low endemicity area of Northern Italy. BMC Infect Dis 2013;13(1):248. 4. Pomares-Estran C, Cenderello G, Ittel A, Karsenti JM, Cardot-Leccia N, VassaloM, Hasseine L, Delaunay P, Rosenthal E, and Marty P: Isolated lymphadenopathy in Leishmania infantum infection: Three case reports. Ann Trop Med Parasitol 2009;103(6):555–559. 5. Mishra S., Shukla A, Tripathi AK, et al.: Visceral leishmaniasis with HIV coinfection and cervical lymphadenopathy. BMJ Case Rep, published online. Accessed May 7, 2014.
Address correspondence to: Giovanni Cenderello Department of Infectious Diseases Galliera Hospital Via Volta 8 Genoa 16128 Italy E-mail: [email protected]
