Treatment of Bolivian mucosal leishmaniasis with miltefosine.

BACKGROUND: Although mucosal leishmaniasis is a prominent disease, it has been studied only to a limited extent. It is classically treated with parenteral antimony or, as a last resort, amphotericin B. METHODS: We treated Bolivian mucosal leishmaniasis due to Leishmania braziliensis with the oral agent miltefosine, 2.5 mg/kg/day for 28 days, and followed-up for 12 months. RESULTS: Seventy-two patients were evaluable. The cure rate for the 36 patients who had "mild" disease (i.e., affecting nasal skin and nasal mucosa) was 83%. The cure rate for the 36 patients who had more extensive disease (involving the palate, pharynx, and larynx) was 58%. Patients refused to be randomized to parenteral agents, but the cure rate for an almost contemporary group who was receiving amphotericin B (45 mg/kg over 90 days) was 7 (50%) of 14. CONCLUSIONS: In this unrandomized trial, oral miltefosine was at least as effective as heroic doses of parenteral amphotericin B. The cure rate for miltefosine was approximately equivalent to historical cure rates using parenteral pentavalent antimony for mild and extensive disease in neighboring Peru. Although gastrointestinal side reactions do occur with miltefosine, its toxicity profile is superior to that of antimony and far superior to that of amphotericin B--in part because of the inherent attractiveness of oral versus parenteral agents. Our results suggest that miltefosine should be the treatment of choice for mucosal disease in North and South America.

Rural campaign to diagnose and treat mucocutaneous leishmaniasis in Bolivia.

Mucocutaneous leishmaniasis (MCL) is endemic in the tropical Amazonian lowlands of Bolivia, an area that regularly receives influxes of migratory populations. In these new agricultural development areas, a campaign to diagnose and treat the disease was carried out between 1989 and 1992, in order to provide direct access to MCL treatment in the endemic areas at a standard equivalent to that offered in the urban centres in Bolivia. The campaign led to the creation of decentralized local centres for diagnosis and treatment of the disease. A campaign to inform the population about leishmaniasis was also undertaken and courses were run to educate medical and paramedical personnel. As a result of the campaign, 3285 cases of leishmaniasis were diagnosed, including 2152 cutaneous and 326 mucosal forms. Also, a total of 1888 cases were treated, 1677 of which cutaneous and 211, mucosal.

Multiple primitive neuroectodermal tumors.

Primitive neuroectodermal tumors are rare, highly aggressive neoplasms that affect both sexes and occur in all age groups. They are a large group of neoplasms with neuroepithelial differentiation, including cutaneous neuroblastomas. Histopathologically they are characterized by a rather uniform population of small, dark cells, with or without Homer Wright rosettes. Immunohistochemically they stain for one or more of the reputed neural markers. Ultrastructurally they show interdigitating processes containing neurosecretory granules, intermediate filaments, and microtubules. We describe a patient with unusual multiple peripheral neuroectodermal tumors localized to the skin, with benign biologic behavior. The patient related the appearance of new lesions for several years without evidence of visceral dissemination or systemic complications. The histopathologic, immunohistochemical, and ultrastructural findings support the diagnosis of a peripheral neuroectodermal tumor.

[Histopathology of mucocutaneous leishmaniasis caused by Leishmania (Vianna) braziliensis]. / Histopathologie de la leishmaniose cutanéo-muqueuse à Leishmania (Viannia) braziliensis.

A histopathological study of mucocutaneous leishmaniasis due to Leishmania (Viannia) braziliensis was carried out on 28 cutaneous and 114 mucosal biopsies, taken from Bolivian and Peruvian patients. This study showed similar histopathological findings in cutaneous and mucosal lesions. The cutaneous biopsies showed a strong epidermal hyperplasia occasionnally budding in the dermis. In the ulcerative area, the epidermis was totally necrosed and replaced by a fibrino-leucocytic edge. In the dermis, histio-lympho-plasmocytic infiltration was constantly found. The histiocytes often gathered in follicles sometimes with diffuse fibrosis. The parasites were encountered in 28.6 p. 100 of the biopsies. Whatever the mucosa concerned (i.e. nasal, palatal or lingual), the mucosal lesion was not different from the cutaneous lesion. The malpighian epithelium is either absent or the seat of a pseudo-epitheliomatous hyperplasia. Major histio-lympho-plasmocytic infiltration was found and extended through the depth of the lamina propria. Suppurative and fibrinoid necroses coexisted superficially and sometimes penetrated in depth. The parasites were found in about 30 p. 100 of the cases.

[Epidemiological, clinical and biological features of mucocutaneous leishmaniasis in Bolivia after a 221 patient sample]. / Particularités épidémiologiques, cliniques et biologiques de la leishmaniose cutanéo-muqueuse en Bolivie d'après un échantillon de 221 malades.

The authors present a clinical, biological and epidemiological study of 221 cases of mucocutaneous leishmaniasis observed in Bolivia between January 1990 and April 1991. The patients exhibited cutaneous lesions in 136 cases and mucous involvement in 85 cases. It is stressed on the severity of the disease in Bolivia.

Electrocardiographic alterations during treatment of mucocutaneous leishmaniasis with meglumine antimoniate and allopurinol.

The electrocardiographic (ECG) changes in Bolivian patients with mucocutaneous leishmaniasis, treated with meglumine antimoniate and allopurinol, were evaluated. Electric changes due to the antimonial compound appeared in 45% of the patients, and consisted of repolarization alteration, principally affecting the T wave and the S-T segment. The changes disappeared within 2 months following the end of the antimonial treatment. In patients with associated Chagas disease and leishmaniasis, antimonial therapy did not aggravate the ECG changes characteristic of Chagasic cardiopathy.

[Eczematide-like purpura (itching purpura--angiodermatitis pruriginosa disseminata)]. / Eczematide-like púrpura (itching púrpura--angiodermitis pruriginosa diseminada).

Between the group of pigmentary and purpury dermatitis, the eczematoid like purpura is better known for its clinical, histological and defined evolutive characteristics. We reviewed all existent biographies and presented two cases of this unusual afection, both with allergic backgrounds, inflammatory capillary lesions and purpura that began in the region of the malleolus and progressed towards the legs, thighs, pelvic girdle, until the chest. These lesions were essentially pruriginous. The histology did not demonstrate signs of allergic vasculitis. What drew our attention was the presence of red blood cells in the Malpighi stratum mucosa. Due to the scarcity of literature and few cases we were not able to clarify satisfactorily this epidermotrophism of the blood cells.

[Epidermotropic reticulosis (Woringer-Kolopp disease)]. / Reticulosis epidermotrópica (enfermedad de Woringer-Kolopp).

The Woringer-Kolopp's disease must be considered a clinical-pathologic variant of mycosis fungoides. It is characterized by heterogenous clinical features and a peculiar histological aspect given by the cellular infiltration of the epidermis. The orgin of the reticular elements is discussed. Though some investigators think they originate in the dermis, others consider their origin to be "in situ". The evolution and prognosis are not well defined, as is the case with the hyperplastic, heteromorphic and polymorphic reticuloses. The case presented has had Hodgkin's disease and then developed a picture of pagetoid reticulosis.