Successful Treatment of Cutaneous Leishmaniasis Caused by Leishmania aethiopica with Liposomal Amphothericin B in an Immunocompromised Traveler Returning from Eritrea

Abstract

Cutaneous leishmaniasis (CL) caused by Leishmania aethiopica is rarely encountered outside disease-endemic areas and there have been no clinical trials evaluating its pharmacotherapy. Under resource limited conditions, antiparasitic treatment of L. aethiopica infection relies largely on pentavalent antimonials. However, treatment failure is frequent and systemic application of these drugs is potentially harmful. [1][2][3][4] Evidence for the efficacy of less adverse chemotherapeutics is needed. We report treatment of CL caused by L. aethiopica using liposomal amphothericin B (LAmB) outside a diseaseendemic region.A 38-year-old man from Eritrea and a permanent resident in Germany over the past 22 years had a 6 × 5 cm skin lesion over his left zygomatic bone. Eleven months earlier, he had returned from a one-year stay in Eritrea where he had visited the coastal area of Asmara and the highlands of the Debub Region. Without an obvious reason, he had stopped taking methotrexate and leflunomid for treatment of rheumatoid arthritis during his stay abroad. Because of increasing inflammation of his left ankle and wrist after returning to Germany, the patient had been treated with infliximab, methotrexate, and prednisolone over the past six months. Two weeks after initiation of this regimen, he recognized a small papule on his left cheek that had gradually progressed to a large lesion seen at the time of hospitalization ( Figure 1A ).Skin biopsy specimens for histologic anlaysis and culture were obtained. Microscopically, a chronic lympho-plasmacellular infiltration with increased content of eosinophilic granulocytes was dominant. Amastigote Leishmania or granulomata were not present. Specific staining results for fungi and mycobacteria were negative, and cultures did not demonstrate Leishmania .However, sequences specific for Old World species could be detected in extracted DNA.5 For species differentiation, we amplified and sequenced the ribosomal internal transcribed spacer region 1 separating the 18S and 5.8S ribosomal RNA genes, which showed infection with L. aethiopica (GenBank accession no. FN252411).6, 7 IgG against recombinant rK39 antigen (immunochromatographic test) and a low positive IgG titer of 1:80 against L. donovani promastigote antigens (indirect immunofluorescence test, cutoff value = 1:40) were detected. An abdominal ultrasound scan, a full blood count, and a polymerase chain reaction for Leishmania spp. from the buffy coat did not suggest visceral disease.We stopped treatment with infliximab and administered 4.4 g LAmB (60 mg/kg body weight) in doses of 200 mg/day for 22 days through a peripheral venous line. Other than mild and transient renal impairment, this therapy was well tolerated. The lesion started to resolve during the third week of LAmB therapy. At discharge, treatment with hydroxychloroquine and sulfasalazine was initiated. However, one month later, this regimen was changed to etanercept, prednisolone, and methotrexate because of increasing rheumatic disease activity. Despite restarting th...

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