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Case report: Successful treatment of cutaneous leishmaniasis caused by Leishmania aethiopica with liposomal amphothericin B in an immunocompromised traveler returning from Eritrea.

ABSTRACT: Cutaneous leishmaniasis caused by Leishmania aethiopica is rarely encountered outside disease-endemic areas and there have been no clinical trials evaluating its pharmacotherapy. We describe the treatment of cutaneous leishmaniasis caused by L. aethiopica using liposomal amphothericin B in an immunocompromised traveler returning from Eritrea.

SUBMITTER: Zanger P

PROVIDER: S-EPMC3083735 | biostudies-literature | 2011 May

REPOSITORIES: biostudies-literature

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Case report: Successful treatment of cutaneous leishmaniasis caused by Leishmania aethiopica with liposomal amphothericin B in an immunocompromised traveler returning from Eritrea.

Zanger Philipp P Kötter Ina I Raible Armin A Gelanew Tesfaye T Schönian Gabriele G Kremsner Peter G PG

The American journal of tropical medicine and hygiene 20110501 5

Cutaneous leishmaniasis caused by Leishmania aethiopica is rarely encountered outside disease-endemic areas and there have been no clinical trials evaluating its pharmacotherapy. We describe the treatment of cutaneous leishmaniasis caused by L. aethiopica using liposomal amphothericin B in an immunocompromised traveler returning from Eritrea. ...[more]

PMID: 21540377

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Project description:Leishmania aethiopica is the etiological agent of cutaneous leishmaniasis (CL) in Ethiopia and can cause severe and complicated cases such as diffuse CL (DCL), mucocutaneous leishmaniasis or extensive CL, requiring systemic treatment. Despite the substantial burden, evidence-based treatment guidelines are lacking. We conducted a systematic review of clinical studies reporting on treatment outcomes of CL due to L aethiopica in order to help identify potentially efficacious medications on CL that can be taken forward for clinical trials. We identified a total of 24 records reporting on 506 treatment episodes of CL presumably due to L aethiopica. The most commonly used drugs were antimonials (n = 201), pentamidine (n = 150) and cryotherapy (n = 103). There were 20 case reports/series, with an overall poor study quality. We only identified two small and/or poor quality randomized controlled trials conducted a long time ago. There were two prospective non-randomized studies reporting on cryotherapy, antimonials and pentamidine. With cryotherapy, cure rates were 60-80%, and 69-85% with antimonials. Pentamidine appeared effective against complicated CL, also in cases non-responsive to antimonials. However, all studies suffered from methodological limitations. Data on miltefosine, paromomycin and liposomal amphotericin B are extremely scarce. Only a few studies are available on DCL. The only potentially effective treatment options for DCL seem to be antimonials with paromomycin in combination or pentamidine, but none have been properly evaluated. In conclusion, the evidence-base for treatment of complicated CL due to L aethiopica is extremely limited. While antimonials remain the most available CL treatment in Ethiopia, their efficacy and safety in CL should be better defined. Most importantly, alternative first line treatments (such as miltefosine or paromomycin) should be explored. High quality trials on CL due to L aethiopica are urgently needed, exploring group sequential methods to evaluate several options in parallel.

Project description:BackgroundLeishmania aethiopica is a unique species that causes cutaneous leishmaniasis (CL), and studies evaluating treatment outcomes for this condition reported inconsistent findings. This study aimed to summarize the evidence on treatment outcomes of CL caused by L. aethiopica to support decisions or propose further study.MethodsWe searched PubMed, Scopus, and ScienceDirect. In addition, we searched grey literature on Google Scholar and performed manual searching on the reference list of articles. Two authors did the screening, selection, critical appraisal, and data extraction. With the narrative synthesis of evidence, we performed a random effects model meta-analysis using the metaprop package in Stata 17. We did sensitivity and subgroup analyses after assessing heterogeneity using the I-squared test and forest plots. The funnel plot and Egger's test were used to assess publication bias.ResultsThe review included 22 studies with 808 participants, and the meta-analysis included seven studies with 677 participants. Most studies documented treatment outcomes with antimonial monotherapy, and only one study reported outcomes with combination therapy. The overall pooled proportion of cure was 63% (95% CI: 38-86%). In the subgroup analysis, systemic antimonial monotherapy showed a cure rate of 61%, and the proportion of cure was 87% with topical therapy. Topical therapy showed a better cure for the localized clinical phenotype. A cohort study documented a cure rate of 94.8% with combination therapy for the localized, mucocutaneous, and diffuse clinical phenotypes. The pooled proportion of unfavourable outcomes was partial response (19%), relapse (17%), discontinuation (19%), and unresponsiveness (6%).ConclusionsThe pooled proportion of cure is low with antimonial monotherapy. Despite limited evidence, combination therapies are a promising treatment option for all clinical phenotypes of CL caused by L. aethiopica. Future high-quality randomized control trials are needed to identify effective monotherapies and evaluate the effectiveness of combination therapies.

Dataset Information

Case report: Successful treatment of cutaneous leishmaniasis caused by Leishmania aethiopica with liposomal amphothericin B in an immunocompromised traveler returning from Eritrea.

Publications

Case report: Successful treatment of cutaneous leishmaniasis caused by Leishmania aethiopica with liposomal amphothericin B in an immunocompromised traveler returning from Eritrea.

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