Clinical observations of unresponsive mucosal leishmaniasis.

Abstract

Abstract. We report the long-term clinical follow-up of two patients with unresponsive mucosal leishmaniasis due to Leishmania (Viannia) braziliensis from the Três Braços area in Bahia State, Brazil. Both were agricultural male workers with extensive upper respiratory mucosal involvement that was not cured with conventional and experimental therapy.In 1985, we reported two male patients with unresponsive mucosal leishmaniasis who appeared to respond to continuous antimony therapy. 1 At that time, we did not realize how long follow-up was necessary to pronounce clinical cure and both patients subsequently relapsed. In 1986, we reported a recurrence in one of these patients. 2 We now report their long-term follow-up to the end of 1996. Given that a parasitologic cure is difficult to prove and may never occur in some forms of leishmaniasis, 3 response to treatment and relapse were established based on clinical criteria of granuloma activity followed by confirmatory biopsy. All parasite isolations identified by monoclonal antibodies and isoenzyme taxonomy have been Leishmania (Viannia) braziliensis. The last clinical evaluation 1996 showed active mucosal granulomata despite all available treatment. This is the justification for the title of this paper. One of us (PDM) had followed these patients for 18 years. Despite recurrences after treatment, recent granulomata seem less aggressive. Both patients do not have any associated disease or condition such as tuberculosis, schistosomiasis, or blastomycosis, that can explain their peculiar evolution, which we have seen complicating recovery from L. (V.) braziliensis infection in humans. These patients lead normal lives and have growing families. They have spent long periods as hospital inpatients and are not anxious to re-enter the hospital. A brief update of their case histories is given with a tabulated summary of treatment regimens. CASE REPORTSPatient MB (LTB12), a 42-year old man, developed mucosal granulomata at the age of 20. He had no history or sign of a cutaneous lesion. The nose, both palates, and the larynx were affected. Leishmania (Viannia) braziliensis was isolated from a nasal mucosal lesion and characterized by isoenzymes and monoclonal antibodies in 1984 and has been reisolated seven times during the last 20 years. He always responded well to antimony therapy only to relapse. After our last report, he relapsed two years later and subsequent treatment is detailed in Table 1. In our previous report, he appeared to have responded to 85 days of therapy with sodium stibogluconate (Pentostam; Wellcome, London, United Kingdom) (20 mg/kg/day) in the hospital to the tolerance limit. 1 In 1996, he had active granulomata around the nasal septal perfuration confirmed by histology. He also had gran-*Deceased. uloma on the right inferior turbinate and a immunofluorescent antibody titer of 1:80. In our laboratory, a titer Ͼ 1:20 is regarded as significant. Mucosal surfaces affected over time include the nasal septum, inferior turbinates, hard and soft palates, orophary...

The site of cutaneous infection influences the immunological response and clinical outcome of hamsters infected with Leishmania panamensis

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The −2518bp promoter polymorphism at CCL2/MCP1 influences susceptibility to mucosal but not localized cutaneous leishmaniasis in Brazil

“…Concomitantly, or months to years later, ~3% of individuals affected by CL develop ML (Carvalho et al, 1985). This severe form is characterized by destructive lesions which may be incapacitating and disfiguring; occasionally becoming life-threatening when lesions of the pharynx and larynx obstruct the respiratory passages and/or cause difficulty in swallowing (Marsden et al, 1998). ML is characterized by a strong cell-mediated immune response with intense infiltrate, pronounced production of inflammatory cytokines such as interferon-γ (IFN-γ) and tumour necrosis factor-α (TNF-α) (Bacellar et al, 2002).…”

The Value of the Otorhinolaryngologic Exam in Correct Mucocutaneous Leishmaniasis Diagnosis

“…3,5 In parallel, histopathologic analysis or anti-Leishmania serology are used for diagnosis of ML. Considering that ML patients do not undergo spontaneous cure and present fatal outcome if left untreated and that ML treatment requires a longer drug regiment than CL, [6][7][8] a proper diagnosis of this particular clinical manifestation is of utmost importance.…”