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Summary: American tegumentary leishmaniasis (ATL) can occur in different forms, classically categorised as cutaneous leishmaniasis, mucosal leishmaniasis, diffuse cutaneous leishmaniasis and disseminated leishmaniasis. We analysed the presence of atypical manifestations (vegetative, verrucous, crusted and lupoid) among a cohort of patients presenting to the Health Post of Corte de Pedra, Bahia, Brazil. Among 1396 patients diagnosed with ATL in 2005–2006, 35 patients (2.5%) presented with atypical manifestations of the disease. Of these patients, 14 were pregnant women, 2 were co-infected with HIV and 19 had no co-morbidity or other apparent risk factors for the development of atypical ATL. The latter 19 patients were the focus of this study. They were predominantly adult males, frequently presenting with facial lesions [P <0.001; odds ratio (OR)=17.5, 95% CI 6.1–52.4] and had higher rates of treatment failure with antimonial therapy (P <0.001; OR=327, 95% CI 45–6668) compared with patients with classic ATL attending in the same period. Thirteen cases healed with amphotericin B, introduced after failure of three or more courses of antimony, suggesting that amphotericin B should be considered as the drug of choice for all patients diagnosed with atypical ATL. [Copyright &y& Elsevier]