Relapsing Visceral Leishmaniasis and Post-Kala-Azar Dermal Leishmaniasis in a Patient with Chronic Inflammatory Demyelinating Polyneuropathy Under Immunosuppression: A Case Report
Articolo
Data di Pubblicazione:
2025
Citazione:
Relapsing Visceral Leishmaniasis and Post-Kala-Azar Dermal
Leishmaniasis in a Patient with Chronic Inflammatory
Demyelinating Polyneuropathy Under Immunosuppression:
A Case Report / Sircana, M.C.F., Bozzi, E., Caturano, A., Cherchi, F., Babudieri, S., Manetti, R.. - (2025).
Abstract:
Visceral leishmaniasis (VL) is a neglected vector-borne disease caused by obligate intracellular protozoa of the genus Leishmania. In immunocompromised patients, VL may present
atypically, progress more aggressively, and respond less favorably to treatment. We present
the case of a 62-year-old male with chronic inflammatory demyelinating polyneuropathy
(CIDP) receiving long-term corticosteroids and azathioprine who developed relapsing VL
complicated by post-kala-azar dermal leishmaniasis (PKDL). The patient initially presented
with prolonged fever, pancytopenia, hepatosplenomegaly, and weight loss. Bone marrow
aspirate revealed Leishmania amastigotes. Intravenous lyposomal amphotericin B (L-AMB)
achieved temporary remission; however, PKDL and VL recurred one year later. Despite
receiving sequential therapy with L-AMB and miltefosine, the patient experienced further
relapses, likely due to severe T- and B-cell lymphopenia and marasmic-like malnutrition.
VL should be considered in the differential diagnosis of prolonged fever and cytopenias
in immunosuppressed patients in Mediterranean Europe, even in the absence of travel
history. Chronic immunosuppression, secondary immunodeficiency, and malnutrition can
significantly impair treatment response and favor recurrence, highlighting the need for
integrated clinical, nutritional, and epidemiological management strategies.
atypically, progress more aggressively, and respond less favorably to treatment. We present
the case of a 62-year-old male with chronic inflammatory demyelinating polyneuropathy
(CIDP) receiving long-term corticosteroids and azathioprine who developed relapsing VL
complicated by post-kala-azar dermal leishmaniasis (PKDL). The patient initially presented
with prolonged fever, pancytopenia, hepatosplenomegaly, and weight loss. Bone marrow
aspirate revealed Leishmania amastigotes. Intravenous lyposomal amphotericin B (L-AMB)
achieved temporary remission; however, PKDL and VL recurred one year later. Despite
receiving sequential therapy with L-AMB and miltefosine, the patient experienced further
relapses, likely due to severe T- and B-cell lymphopenia and marasmic-like malnutrition.
VL should be considered in the differential diagnosis of prolonged fever and cytopenias
in immunosuppressed patients in Mediterranean Europe, even in the absence of travel
history. Chronic immunosuppression, secondary immunodeficiency, and malnutrition can
significantly impair treatment response and favor recurrence, highlighting the need for
integrated clinical, nutritional, and epidemiological management strategies.
Tipologia CRIS:
1.1 Articolo in rivista
Keywords:
visceral leishmaniasis, post-kala-azar dermal leishmaniasis, secondary
immunodeficiency, CIDP, malnutrition, amphotericin B, miltefosine
Elenco autori:
Sircana, Marta Chiara Francesca; Bozzi, Elena; Caturano, Alfredo; Cherchi, Francesca; Babudieri, Sergio; Manetti, Roberto
Link alla scheda completa: