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Alveolar echinococcosis (AE) is an important parasitic infestation caused by the larval stage of the Echinococcus multilocularis. Turkey is one of the countries occurred endemic of the disease. The disease primarily affects the liver. Cerebral and craniofacial involvements are very rare. Here the authors report a 46-year-old woman with simultaneous cerebral and craniofacial involvement of E. multilocularis. Although primary AE lesions with simultaneous cerebral and craniofacial involvement are exceedingly rare, radiologist must suspect from other unusual involvement such as craniofacial and muscular location in patients with cerebral AE.