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Refractory mucocutaneous leishmaniasis resolved with combination treatment based on intravenous pentamidine, oral azole, aerosolized liposomal amphotericin B, and intralesional meglumine antimoniate.
Int J Infect Dis. 2020 Aug; 97:204-207.IJ

Abstract

INTRODUCTION

Mucocutaneous leishmaniasis (MCL) is a complication of tegumentary leishmaniasis, causing potentially life-threatening lesions in the ear, nose, and throat (ENT) region, and most commonly due to Leishmania (Viannia) braziliensis. We report a case of relapsing MCL in an Italian traveler returning from Argentina.

CASE DESCRIPTION

A 65-year-old Italian male patient with chronic kidney disease, arterial hypertension, prostatic hypertrophy, and type-2 diabetes mellitus was referred for severe relapsing MCL acquired in Argentina. ENT examination showed severe diffuse pharyngolaryngeal edema and erythema, partially obstructing the airways. A nasopharyngeal biopsy revealed a lymphoplasmacytic inflammation and presence of Leishmania amastigotes, subsequently identified as L. (V.) braziliensis by hsp70 PCR-RFLP analysis and sequencing. Despite receiving four courses of liposomal amphotericine B (L-AmB) and two courses of miltefosine over a 2-year period, the patient presented recurrence of symptoms a few months after the end of each course. After the patient was referred to us, a combined treatment was started with intravenous pentamidine 4 mg/kg on alternate days for 10 doses, followed by one dose per week for an additional seven doses, intralesional meglumine antimoniate on the nasal lesion once per week for six doses, oral azoles for three months, and aerosolized L-AmB on alternate days for three months. The treatment led to regression of mucosal lesions and respiratory symptoms. Renal function temporarily worsened, and the addition of insulin was required to maintain glycemic compensation after pentamidine discontinuation.

CONCLUSIONS

This case highlights the difficulties in managing a life-threatening refractory case of MCL in an Italian traveler with multiple comorbidities. Even though parenteral antimonial derivatives are traditionally considered the treatment of choice for MCL, they are relatively contraindicated in cases of chronic kidney disease.The required dose adjustment in cases of impaired renal function is unknown, therefore the use of alternative drugs is recommended. This case was resolved with combination treatment, including aerosolized L-AmB, which had never been used before for MCL.

Authors+Show Affiliations

Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy.Otorhinolaryngology Unit, Careggi University Hospital, Florence, Italy.Otorhinolaryngology Unit, Careggi University Hospital, Florence, Italy.Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy.Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy.Infectious and Tropical Diseases Unit, Careggi University Hospital, Florence, Italy.Otorhinolaryngology Unit, Careggi University Hospital, Florence, Italy.Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy; Otorhinolaryngology Unit, Careggi University Hospital, Florence, Italy.Department of Health Sciences, Clinical Pharmacology and Oncology Section, University of Florence, Florence, Italy.Unit of Vector-borne Diseases, Department of Infectious Diseases, Istituto Superiore di Sanità, Rome, Italy.Unit of Vector-borne Diseases, Department of Infectious Diseases, Istituto Superiore di Sanità, Rome, Italy.Unit of Vector-borne Diseases, Department of Infectious Diseases, Istituto Superiore di Sanità, Rome, Italy.Clinic of Infectious Diseases, Vita-Salute San Raffaele University, Milan, Italy.Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy; Infectious and Tropical Diseases Unit, Careggi University Hospital, Florence, Italy; Referral Center for Tropical Diseases of Tuscany, Infectious and Tropical Diseases Unit, Careggi University Hospital, Florence, Italy.Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy; Infectious and Tropical Diseases Unit, Careggi University Hospital, Florence, Italy; Referral Center for Tropical Diseases of Tuscany, Infectious and Tropical Diseases Unit, Careggi University Hospital, Florence, Italy. Electronic address: lorenzo.zammarchi@unifi.it.

Pub Type(s)

Case Reports

Language

eng

PubMed ID

32505874

Citation

Basile, Gregorio, et al. "Refractory Mucocutaneous Leishmaniasis Resolved With Combination Treatment Based On Intravenous Pentamidine, Oral Azole, Aerosolized Liposomal Amphotericin B, and Intralesional Meglumine Antimoniate." International Journal of Infectious Diseases : IJID : Official Publication of the International Society for Infectious Diseases, vol. 97, 2020, pp. 204-207.
Basile G, Cristofaro G, Locatello LG, et al. Refractory mucocutaneous leishmaniasis resolved with combination treatment based on intravenous pentamidine, oral azole, aerosolized liposomal amphotericin B, and intralesional meglumine antimoniate. Int J Infect Dis. 2020;97:204-207.
Basile, G., Cristofaro, G., Locatello, L. G., Vellere, I., Piccica, M., Bresci, S., Maggiore, G., Gallo, O., Novelli, A., Di Muccio, T., Gramiccia, M., Gradoni, L., Gaiera, G., Bartoloni, A., & Zammarchi, L. (2020). Refractory mucocutaneous leishmaniasis resolved with combination treatment based on intravenous pentamidine, oral azole, aerosolized liposomal amphotericin B, and intralesional meglumine antimoniate. International Journal of Infectious Diseases : IJID : Official Publication of the International Society for Infectious Diseases, 97, 204-207. https://doi.org/10.1016/j.ijid.2020.06.003
Basile G, et al. Refractory Mucocutaneous Leishmaniasis Resolved With Combination Treatment Based On Intravenous Pentamidine, Oral Azole, Aerosolized Liposomal Amphotericin B, and Intralesional Meglumine Antimoniate. Int J Infect Dis. 2020;97:204-207. PubMed PMID: 32505874.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Refractory mucocutaneous leishmaniasis resolved with combination treatment based on intravenous pentamidine, oral azole, aerosolized liposomal amphotericin B, and intralesional meglumine antimoniate. AU - Basile,Gregorio, AU - Cristofaro,Glauco, AU - Locatello,Luca Giovanni, AU - Vellere,Iacopo, AU - Piccica,Matteo, AU - Bresci,Silvia, AU - Maggiore,Giandomenico, AU - Gallo,Oreste, AU - Novelli,Andrea, AU - Di Muccio,Trentina, AU - Gramiccia,Marina, AU - Gradoni,Luigi, AU - Gaiera,Giovanni, AU - Bartoloni,Alessandro, AU - Zammarchi,Lorenzo, Y1 - 2020/06/04/ PY - 2020/03/25/received PY - 2020/05/29/revised PY - 2020/06/01/accepted PY - 2020/6/9/pubmed PY - 2020/10/6/medline PY - 2020/6/8/entrez KW - Aerosolized liposomal amphotericin B KW - Chronic kidney disease KW - Combination therapy KW - Mucocutaneous leishmaniasis KW - Pentamidine KW - Recurrent leishmaniasis SP - 204 EP - 207 JF - International journal of infectious diseases : IJID : official publication of the International Society for Infectious Diseases JO - Int J Infect Dis VL - 97 N2 - INTRODUCTION: Mucocutaneous leishmaniasis (MCL) is a complication of tegumentary leishmaniasis, causing potentially life-threatening lesions in the ear, nose, and throat (ENT) region, and most commonly due to Leishmania (Viannia) braziliensis. We report a case of relapsing MCL in an Italian traveler returning from Argentina. CASE DESCRIPTION: A 65-year-old Italian male patient with chronic kidney disease, arterial hypertension, prostatic hypertrophy, and type-2 diabetes mellitus was referred for severe relapsing MCL acquired in Argentina. ENT examination showed severe diffuse pharyngolaryngeal edema and erythema, partially obstructing the airways. A nasopharyngeal biopsy revealed a lymphoplasmacytic inflammation and presence of Leishmania amastigotes, subsequently identified as L. (V.) braziliensis by hsp70 PCR-RFLP analysis and sequencing. Despite receiving four courses of liposomal amphotericine B (L-AmB) and two courses of miltefosine over a 2-year period, the patient presented recurrence of symptoms a few months after the end of each course. After the patient was referred to us, a combined treatment was started with intravenous pentamidine 4 mg/kg on alternate days for 10 doses, followed by one dose per week for an additional seven doses, intralesional meglumine antimoniate on the nasal lesion once per week for six doses, oral azoles for three months, and aerosolized L-AmB on alternate days for three months. The treatment led to regression of mucosal lesions and respiratory symptoms. Renal function temporarily worsened, and the addition of insulin was required to maintain glycemic compensation after pentamidine discontinuation. CONCLUSIONS: This case highlights the difficulties in managing a life-threatening refractory case of MCL in an Italian traveler with multiple comorbidities. Even though parenteral antimonial derivatives are traditionally considered the treatment of choice for MCL, they are relatively contraindicated in cases of chronic kidney disease.The required dose adjustment in cases of impaired renal function is unknown, therefore the use of alternative drugs is recommended. This case was resolved with combination treatment, including aerosolized L-AmB, which had never been used before for MCL. SN - 1878-3511 UR - https://www.unboundmedicine.com/medline/citation/32505874/Refractory_mucocutaneous_leishmaniasis_resolved_with_combination_treatment_based_on_intravenous_pentamidine_oral_azole_aerosolized_liposomal_amphotericin_B_and_intralesional_meglumine_antimoniate_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S1201-9712(20)30438-0 DB - PRIME DP - Unbound Medicine ER -