An ethnobotanical and ethnopharmacognostic survey has been carried out in one of the most isolated mountainous area in Europe: the village of Lepushe and its surrounding territory, in the Northern Albanian Alps. Approximately 70 botanical taxa and 160 preparations, mainly derived from plants, but also derived from animal products or minerals, have been recorded. The archaic belief of the signature still plays a very important role in the present ethnomedicine of the Albanians of Lepushe. As a consequence, aerial parts of Chelidonium majus are used to treat jaundice; leaves of the fern Phyllitis scolopendrium are thought to be able to treat every respiratory and lung affection; the bulbs of Lilium martagon are used to treat liver diseases; jasper is rubbed into milk and given to sheep to drink and, the membrane of a hen's muscular stomach is used to treat human kidney stones. A current common ethnomedical use of P. scolopendrium and L. martagon is recorded for the first time in Europe.