Background Montenegro is an endemic area for visceral leishmaniasis. Natural condition and geographical position (Mediterranean area) allow of visceral leishmaniasis existence. Epidemiological studies warn on the increase in number of disease in Montenegro, which are 3 patients on 646000 inhabitants per year. Methods The investigations used epidemiological, clinical and laboratory methods. Diagnosis was etiologically confirmed through bone marrow bioptate analysis, by direct microscoping of serial sections colored with the Giemsa s stain, Reticulin (Gordon and Sweet method), PAS method, and by immune-biochemical methods, and serological method. Results In the period from 1992 to 2011 in Montenegro, 67 cases of leishmaniasis have been diagnosed, with 4 lethal outcomes. The children population participate with 36 (%) cases, age 0-4 years in 22 (40%) and age 5-15 years in 14 (%). Which the prevalence of general infective syndrome (high febricity, exhaustion), in all children cases, enlarged of spleen in 28 cases, and liver in 26 cases, anemia in 29 cases, pancytopenia in 22 cases, increased serum transaminases in 27 cases. Recidives were registered in 9 patients. According to the geographic area where the disease was diagnosed the expansion include the entire coastal area of Montenegro from Ulcinj to Herceg Novi, Cetinje, the area of Skadar lake including Podgorica and even some northern parts of Montenegro. Conclusion Expansion of the primary endemic focus, more severe types of the disease, co-infective types and increase in mortality, therapy resistance, coinfective forms of disease are the new features of leishmaniasis in Montenegro.