Diffuse intralobular fibrosis of the liver is rare in cases of New World visceral leishmaniasis. A patient with this disease from a newly endemic area in the northernmost area of the Brazilian Amazon region was studied. Hypertrophy and hyperplasia of the phagocytic mononuclear cell system were observed, with parasitism and Disse's space fibrosis diffusely involving the liver. This description is based on a biopsy carried out after seven days of treatment with Glucantime®. Another biopsy performed almost two years later showed no fibrosis, no Kupffer cell hypertrophy and hyperplasia, and no parasitism. The mechanism of fibrosis and regression is discussed.