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Atypical Presentation of PKDL due to Leishmania infantum in an HIV-Infected Patient with Relapsing Visceral Leishmaniasis.

Celesia BM, Cacopardo B, Massimino D, Gussio M, Tosto S, Nunnari G, Pinzone MR - Case Rep Infect Dis (2014)

Bottom Line: At the time of PKDL appearance, the patient was virologically suppressed but had failed to restore an adequate CD4+ T-cell count.Paradoxically, cutaneous lesions markedly improved when a new relapse of VL occurred.The therapeutic management of both PKDL and VL in HIV infection is challenging, because relapses are frequent and evidence is often limited to small case series and case reports.

View Article: PubMed Central - PubMed

Affiliation: Department of Clinical and Molecular Biomedicine, Division of Infectious Diseases, University of Catania, Via Palermo 636, 95125 Catania, Italy.

ABSTRACT
We describe the case of an Italian patient with HIV infection who developed an atypical rash resembling post-kala-azar dermal leishmaniasis (PKDL) when receiving liposomal Amphotericin B (L-AMB) for secondary prophylaxis of visceral leishmaniasis (VL). At the time of PKDL appearance, the patient was virologically suppressed but had failed to restore an adequate CD4+ T-cell count. Histology of skin lesions revealed the presence of a granulomatous infiltrate, with lymphocytes, plasma cells, and macrophages, most of which contained Leishmania amastigotes. Restriction fragment length polymorphism-polymerase chain reaction was positive for Leishmania infantum. Paradoxically, cutaneous lesions markedly improved when a new relapse of VL occurred. The patient received meglumine antimoniate, with a rapid clinical response and complete disappearance of cutaneous rash. Unfortunately, the patient had several relapses of VL over the following years, though the interval between them has become wider after restarting maintenance therapy with L-AMB 4 mg/kg/day once a month. Even if rare, PKDL due to Leishmania infantum may occur in Western countries and represents a diagnostic and therapeutic challenge for physicians. The therapeutic management of both PKDL and VL in HIV infection is challenging, because relapses are frequent and evidence is often limited to small case series and case reports.

No MeSH data available.


Related in: MedlinePlus

Diffuse maculopapular and nodular rash on the upper limbs (a) and trunk (b).
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fig1: Diffuse maculopapular and nodular rash on the upper limbs (a) and trunk (b).

Mentions: In May 2002, a 33-year-old Italian man presented to our outpatient clinic complaining of fever, weight loss, and diarrhea. He had mucocutaneous lesions highly suggestive of Kaposi's sarcoma (KS). On physical examination he had hepatosplenomegaly; laboratory tests showed pancytopenia and polyclonal hypergammaglobulinemia. He tested HIV positive. His CD4+ T-cell count was 17 cells/μL and HIV RNA viral load was 225,181 copies/mL. A bone marrow smear showed the presence of Leishmania spp. amastigotes. As a consequence, diagnosis of VL was made and treatment with meglumine antimoniate at a dose of 20 mg/kg/day was given for 30 days. Moreover, HAART with Lamivudine-Stavudine-Lopinavir/ritonavir was started. The patient clinically improved, with progressive resolution of KS lesions and good virological response to antiretroviral therapy. Six months later, the patient suffered a clinical relapse of VL and was treated with intravenous liposomal Amphotericin B (L-AMB) at a dose of 4 mg/kg/day for 5 days and then weekly at the same dose for 4 weeks. However, several relapses of VL occurred over the following years. Each relapse was treated with the aforementioned drug schedule. In February 2008, it was decided to start maintenance therapy with L-AMB at a dose of 4 mg/kg/day once a month, in an attempt to prevent further relapses. At this time, his HAART regimen consisted of Abacavir-Lamivudine-Lopinavir/ritonavir; HIV viral load was stably below 50 copies/mL and CD4+ T-cell count was 125 cells/μL. Some weeks after starting secondary prophylaxis with L-AMB, a maculopapular and nodular rash appeared on his trunk and upper limbs (Figure 1). The rash gradually worsened and, four months later, treatment with L-AMB was stopped and a cutaneous biopsy was performed. Histology revealed the presence of a granulomatous infiltrate, with lymphocytes, plasma cells, and macrophages, most of which contained Leishmania amastigotes. Restriction fragment length polymorphism-polymerase chain reaction (RFLP-PCR) was positive for L. infantum. These findings were consistent with a diagnosis of PKDL. In September 2008, the patient started complaining of fever, diarrhea, and night sweats. Paradoxically, cutaneous lesions markedly improved. On the basis of his symptoms, a relapse of VL was suspected and confirmed by bone marrow aspirate, which showed the presence of Leishmania amastigotes. Treatment with meglumine antimoniate was restarted at a dose of 20 mg/kg/day for 30 days. The patient experienced a rapid clinical response and complete disappearance of cutaneous rash. Unfortunately, the patient had several relapses of VL over the following years, though the interval between them has become wider after restarting maintenance therapy with L-AMB 4 mg/kg/day once a month. At the last followup in December 2013, the patient was in good health and was still receiving secondary prophylaxis with L-AMB. He was taking HAART with Abacavir-Lamivudine-Lopinavir/ritonavir-raltegravir with his last CD4+ T-cell count being 98 cells/μL and HIV RNA < 20 copies/mL.


Atypical Presentation of PKDL due to Leishmania infantum in an HIV-Infected Patient with Relapsing Visceral Leishmaniasis.

Celesia BM, Cacopardo B, Massimino D, Gussio M, Tosto S, Nunnari G, Pinzone MR - Case Rep Infect Dis (2014)

Diffuse maculopapular and nodular rash on the upper limbs (a) and trunk (b).
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4150412&req=5

fig1: Diffuse maculopapular and nodular rash on the upper limbs (a) and trunk (b).
Mentions: In May 2002, a 33-year-old Italian man presented to our outpatient clinic complaining of fever, weight loss, and diarrhea. He had mucocutaneous lesions highly suggestive of Kaposi's sarcoma (KS). On physical examination he had hepatosplenomegaly; laboratory tests showed pancytopenia and polyclonal hypergammaglobulinemia. He tested HIV positive. His CD4+ T-cell count was 17 cells/μL and HIV RNA viral load was 225,181 copies/mL. A bone marrow smear showed the presence of Leishmania spp. amastigotes. As a consequence, diagnosis of VL was made and treatment with meglumine antimoniate at a dose of 20 mg/kg/day was given for 30 days. Moreover, HAART with Lamivudine-Stavudine-Lopinavir/ritonavir was started. The patient clinically improved, with progressive resolution of KS lesions and good virological response to antiretroviral therapy. Six months later, the patient suffered a clinical relapse of VL and was treated with intravenous liposomal Amphotericin B (L-AMB) at a dose of 4 mg/kg/day for 5 days and then weekly at the same dose for 4 weeks. However, several relapses of VL occurred over the following years. Each relapse was treated with the aforementioned drug schedule. In February 2008, it was decided to start maintenance therapy with L-AMB at a dose of 4 mg/kg/day once a month, in an attempt to prevent further relapses. At this time, his HAART regimen consisted of Abacavir-Lamivudine-Lopinavir/ritonavir; HIV viral load was stably below 50 copies/mL and CD4+ T-cell count was 125 cells/μL. Some weeks after starting secondary prophylaxis with L-AMB, a maculopapular and nodular rash appeared on his trunk and upper limbs (Figure 1). The rash gradually worsened and, four months later, treatment with L-AMB was stopped and a cutaneous biopsy was performed. Histology revealed the presence of a granulomatous infiltrate, with lymphocytes, plasma cells, and macrophages, most of which contained Leishmania amastigotes. Restriction fragment length polymorphism-polymerase chain reaction (RFLP-PCR) was positive for L. infantum. These findings were consistent with a diagnosis of PKDL. In September 2008, the patient started complaining of fever, diarrhea, and night sweats. Paradoxically, cutaneous lesions markedly improved. On the basis of his symptoms, a relapse of VL was suspected and confirmed by bone marrow aspirate, which showed the presence of Leishmania amastigotes. Treatment with meglumine antimoniate was restarted at a dose of 20 mg/kg/day for 30 days. The patient experienced a rapid clinical response and complete disappearance of cutaneous rash. Unfortunately, the patient had several relapses of VL over the following years, though the interval between them has become wider after restarting maintenance therapy with L-AMB 4 mg/kg/day once a month. At the last followup in December 2013, the patient was in good health and was still receiving secondary prophylaxis with L-AMB. He was taking HAART with Abacavir-Lamivudine-Lopinavir/ritonavir-raltegravir with his last CD4+ T-cell count being 98 cells/μL and HIV RNA < 20 copies/mL.

Bottom Line: At the time of PKDL appearance, the patient was virologically suppressed but had failed to restore an adequate CD4+ T-cell count.Paradoxically, cutaneous lesions markedly improved when a new relapse of VL occurred.The therapeutic management of both PKDL and VL in HIV infection is challenging, because relapses are frequent and evidence is often limited to small case series and case reports.

View Article: PubMed Central - PubMed

Affiliation: Department of Clinical and Molecular Biomedicine, Division of Infectious Diseases, University of Catania, Via Palermo 636, 95125 Catania, Italy.

ABSTRACT
We describe the case of an Italian patient with HIV infection who developed an atypical rash resembling post-kala-azar dermal leishmaniasis (PKDL) when receiving liposomal Amphotericin B (L-AMB) for secondary prophylaxis of visceral leishmaniasis (VL). At the time of PKDL appearance, the patient was virologically suppressed but had failed to restore an adequate CD4+ T-cell count. Histology of skin lesions revealed the presence of a granulomatous infiltrate, with lymphocytes, plasma cells, and macrophages, most of which contained Leishmania amastigotes. Restriction fragment length polymorphism-polymerase chain reaction was positive for Leishmania infantum. Paradoxically, cutaneous lesions markedly improved when a new relapse of VL occurred. The patient received meglumine antimoniate, with a rapid clinical response and complete disappearance of cutaneous rash. Unfortunately, the patient had several relapses of VL over the following years, though the interval between them has become wider after restarting maintenance therapy with L-AMB 4 mg/kg/day once a month. Even if rare, PKDL due to Leishmania infantum may occur in Western countries and represents a diagnostic and therapeutic challenge for physicians. The therapeutic management of both PKDL and VL in HIV infection is challenging, because relapses are frequent and evidence is often limited to small case series and case reports.

No MeSH data available.


Related in: MedlinePlus