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      Good response to pentamidine isethionate in a case of Mucosal Leishmaniasis caused by Leishmania (Viannia) braziliensis that was difficult to treat: Case Report

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          Abstract

          Abstract In Brazil, meglumine antimoniate is the first drug of choice for mucosal leishmaniasis treatment followed by amphotericin B and pentamidine isethionate. We report the case of a patient with severe mucosal lesions caused by Leishmania (Viannia) braziliensis that were difficult to treat. Over a 14-year period, the patient showed low adherence and three treatment attempts with meglumine antimoniate failed. Additionally, there was an unsatisfactory response to liposomal amphotericin B and nephrotoxicity when using amphotericin B deoxycholate that persisted after new treatment attempt with liposomal amphotericin B. Finally, healing was achieved with pentamidine isethionate and maintained during nine months of monitoring.

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          Sensitivity of Leishmania braziliensis promastigotes to meglumine antimoniate (glucantime) is higher than that of other Leishmania species and correlates with response to therapy in American tegumentary leishmaniasis.

          The first line drugs for the treatment of leishmaniasis are antimonial derivatives. Poor clinical response may be credited to factors linked to the host, the drug, or the parasite. We determined the sensitivity of Leishmania sp. promastigotes and amastigotes by counting parasites exposed to increasing concentrations of meglumine antimoniate (Glucantime). Leishmania braziliensis promastigotes were significantly more sensitive than those belonging to other species. The sensitivity of L. braziliensis isolates from patients with unfavorable clinical outcome, such as therapeutic failure or relapse, was significantly lower than those from patients who had clinical cure. Poor clinical response to therapy (therapeutic failure or relapse) was also associated with inadequate antimonial therapy. We also found a significant and positive correlation between promastigotes and intracellular amastigotes with regard to their in vitro susceptibilities to meglumine antimoniate. Our data provide evidence for an association between the sensitivity of promastigotes to antimonials in vitro and clinical response to therapy in American tegumentary leishmaniasis. The high sensitivity of the local L. braziliensis to meglumine antimoniate in vitro provides an explanation for the good clinical response of cutaneous leishmaniasis in the municipality of Rio de Janeiro, Brazil, even when low-dose regimens are employed.
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            Fatores associados ao insucesso do tratamento da leishmaniose cutânea com antimoniato de meglumina

            Foram investigados os fatores associados ao insucesso do tratamento da leishmaniose cutânea com antimoniato de meglumina num serviço de referência para leishmanioses, em Mato Grosso. Uma coorte histórica de 151 pacientes com diagnóstico de leishmaniose cutânea foi construída com informações dos prontuários. A incidência de insucesso após o primeiro ciclo de antimonial foi 47% (IC95%=39,2%-55%). Dose de antimonial inferior a 10mg/kg/dia (RR=1,8; IC95:1,1-3,0), tratamento prévio para leishmaniose (RR=1,7; IC95:1,3-2,4), três ou mais lesões (RR=1,9; IC95:1,4-2,5), tratamento irregular (RR=1,9; IC95:1,3-2,6) e peso maior que 68kg (RR=1,7; IC95:1,1-2,5) foram associados ao insucesso terapêutico. Após ajuste, permaneceram associados ao insucesso os seguintes fatores: 3 ou mais lesões cutâneas (OR=4,6; IC95%=1,2-17,4), tratamento anterior para leishmaniose tegumentar americana (OR=4,5; IC95%=1,1-7,5), peso maior que 68kg (OR=4,3; IC95%=1,5-11,9) e irregularidade no tratamento (OR=12,5; IC95%=2,1-75,4), embora o peso possivelmente tenha sido associado ao insucesso devido à limitação da dose máxima. Estes achados auxiliam na identificação de pacientes com maior risco de insucesso no tratamento da leishmaniose cutânea com antimonial.
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              Mucosal leishmaniasis ("espundia") responsive to low dose of N-methyl glucamine (Glucantime ®) in Rio de Janeiro, Brazil

              Response to treatment with antimonial drugs varies considerably depending on the parasite strain involved, immune status of the patient and clinical form of the disease. Therapeutic regimens with this first line drug have been frequently modified both, in dose and duration of therapy. A regimen of 20 mg/kg/day of pentavalent antimony (Sb5+) during four weeks without an upper limit on the daily dose is currently recommended for mucosal disease ("espundia"). Side-effects with this dose are more marked in elderly patients, more commonly affected by this form of leishmaniasis. According to our experience, leishmaniasis in Rio de Janeiro responds well to antimony and, in cutaneous disease, high cure rates are obtained with 5 mg/kg/day of Sb5+ during 30 to 45-days. In this study a high rate of cure (91.4%) employing this dose was achieved in 36 patients with mild disease in this same geographic region. Side-effects were reduced and no antimony refractoriness was noted with subsequent use of larger dose in patients that failed to respond to initial schedule.
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                Author and article information

                Contributors
                Role: ND
                Role: ND
                Role: ND
                Role: ND
                Role: ND
                Role: ND
                Role: ND
                Role: ND
                Role: ND
                Journal
                rsbmt
                Revista da Sociedade Brasileira de Medicina Tropical
                Rev. Soc. Bras. Med. Trop.
                Sociedade Brasileira de Medicina Tropical - SBMT (Uberaba, MG, Brazil )
                0037-8682
                1678-9849
                January 2019
                : 52
                : 0
                : e-20180236
                Affiliations
                [1] Rio de Janeiro orgnameFundação Oswaldo Cruz orgdiv1Instituto Nacional de Infectologia Brazil
                [2] Rio de Janeiro Rio de Janeiro orgnameUniversidade Federal do Rio de Janeiro orgdiv1Departamento de Otorrinolaringologia e Oftalmologia Brazil
                Article
                S0037-86822019000100700
                10.1590/0037-8682-0236-2018
                3f890566-827d-402f-b9dd-c48f0bbd5863

                This work is licensed under a Creative Commons Attribution 4.0 International License.

                History
                : 16 June 2018
                : 07 August 2018
                Page count
                Figures: 0, Tables: 0, Equations: 0, References: 12, Pages: 0
                Product

                SciELO Brazil

                Categories
                Case Reports

                Mucosal leishmaniasis,Pentamidine isethionate,Therapy

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