Intramedullary spinal cord metastases (ISCMs) represent a rare complication of malignancies and have been identified in 0.9-2.1% of all autopsy cases of cancer [1][2][3]. Lung cancer and breast carcinoma represent the majority of primary tumors (59% and 14%, respectively) [4], and the reported incidence is estimated at 0.1-0.4% [4,5]. They become clinically evident in less than 1% of patients suffering from metastatic disease [6,7]. Most of them present with rapid progressive neurological deficits and require immediate evaluation [2,3,6,8,9]. Nowadays, spinal magnetic resonance imaging (MRI) is routinely applied for diagnosis.Surgery aims at decompression of functional neural tissue and histological confirmation of the tumor [9]. Metastatic localization of a malignant disease is usually solitary and at clinical onset the patient has systemic widespread with secondary localization to lung, liver, bones and brain [10]. In this report we describe the case of a 49 year-old woman, affected by breast cancer, we have operated on for two adjacent cervical ISCMs. To our knowledge, this is the first surgical case reported in literature of a double intramedullary metastatic disease, with the two separate lesions occurring simultaneously. The aim of our paper is to describe the clinical features and radiological findings of metastatic intramedullary breast carcinoma, as well as surgical technique with emphasis on intraoperative neurophysiological monitoring (IONM) and a wide literature review about ISCMs from breast cancer.