Department of Infectious and Parasitic Diseases, Hospital of Clinics, School of Medicine, University of São Paulo, São Paulo, Brazil
Sir,
We describe here a patient infected with the human immunodeficiency virus (HIV) suffering from mucocutaneous leishmaniasis (MCL), treated successfully with liposomal amphotericin B (AmBisome), after having failed with antimonial treatment, and having developed diabetes mellitus with pentamidine, which had to be withdrawn.
The patient, a 35-year-old male, was hospitalized in April 1999, complaining of dysphagia for 1 year. He gave a history of an ulcerated lesion on the leg, which healed spontaneously 20 years ago. He started feeling oral pain in 1987, and was diagnosed as having mucosal leishmaniasis of the palate. The patient was treated with various cycles of N-methyl glucamine antimoniate at a dose of 20 mg/kg/Sbv/day, with reported improvement, without full resolution of signs and symptoms. Four months before admission, a test for HIV (ELISA and Western blot), was positive, and he had observed the appearance of an ulcer on the penis 3 months before admission. Upon admission, physical examination revealed granulomatous and ulcerated lesions on the soft palate, and fibroscopy demonstrated the involvement of larynx and epiglottis, with infiltration in ventricular bands, infiltration of arytenoid regions and moderate reduction of respiratory slit calibre. Laboratory tests showed: a CD4 count of 387 cells/mm3, a CD8 count of 312 cells/mm3, a plasma HIV RNA load of 1.6 x 104 copies/L), Montenegro reaction of 6 mm (positive) and serological reaction for leishmaniasis by indirect immunofluorescence of 1/640. Histological examination of palate and larynx biopsies revealed a chronic granulomatous inflammatory process with a negative immunohistochemical reaction and negative tests for the presence of mycobacteria and fungi. A biopsy of the penis lesion revealed the presence of amastigote leishmania forms. The patient also had hepatosplenomegaly and a liver biopsy showed cirrhosis with discrete inflammatory activity attributed to hepatitis B virus. A search for leishmania and its antigens by immunohistochemical reaction in liver tissue was negative. Treatment of MCL with a total dose of 2400 mg of pentamidine led to healing of the penis ulcer, but the oral lesions persisted and the patient progressed to diabetes mellitus, leading to discontinuation of the drug. Liposomal amphotericin B was introduced at a dose of 3 mg/kg/day, with healing of oropharyngeal and laryngeal lesions after a total dose of 12800 mg, with no side effects during the use of this medication. The patient has been followed up for 5 months, with no reactivation of the lesions.
In Brazil, MCL is caused by Leishmania (Viannia) braziliensis, and is characterized by skin ulcers that progress to spontaneous healing, with dissemination to the mucosae possibly occurring months or decades later.1 Pentavalent antimonials represent the first-choice treatment option, but treatment failure or interruption due to their side effects often occurs.2 Therapeutic alternatives such as conventional amphotericin B and pentamidine isothionate present considerable toxicity.3,4 The number of case reports of MCL and HIV co-infection has progressively increased. MCL may be severe in HIV-infected patients, with possible dissemination and failure of treatment with antimonials and other drugs.5 Our patient presented with signs and symptoms of dissemination, since a skin lesion appeared after involvement of the mucosae, an unusual behaviour in MCL. It is unlikely that the penis ulcer was the result of reinfection because the patient had left the endemic region of leishmaniasis 17 years before. Also, skin lesions habitually occur on exposed regions such as arms, legs or the face. Liposomal amphotericin B has been used previously with success in a small number of cases of mucosal leishmaniasis not associated with HIV that did not respond to antimonials, with the average dose for lesion healing being 3333 mg (ranging from 2000 mg to 5300 mg).6 Our patient required a higher cumulative dose, probably because of the HIV co-infection.
In conclusion, we have described a case of MCL in a patient infected with HIV, and based on a literature survey, we believe that this case is the first report of the use of this medication for the treatment of MCL and HIV co-infection. Liposomal amphotericin B seems to be promising for the treatment of HIV and MCL co-infection, but studies on larger series are needed to confirm its efficacy.
Notes
J Antimicrob Chemother 2000; 46: 341342
* Corresponding author. Tel: +55-11-881-8144; Fax: +55-11-881-8158; E-mail: valdirsa{at}netpoint.com.br
References
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