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Multiple myeloma (MM) is the second most common hematological malignancy (13%), accounting for 1% of all neoplasms, and is characterized by neoplastic proliferation of plasma cells (1). MM is usually confined to the bone marrow, with 80–90% of MM patients exhibiting skeletal involvement. Extramedullary spread is relatively rare; the incidence is only 3–5% (2). Central nervous system involvement in multiple myeloma (CNS-MM) is an uncommon condition developing in 1% of MM patients. CNS-MM is diagnosed by detection of monoclonal plasma cells in the cerebrospinal fluid (CSF) or CNS involvement evident on neuroimaging of previously diagnosed MM patients (3). CNS-MM may manifest as a dural, parenchymal or leptomeningeal lesion (4, 5). However, only a single case of CNS-MM manifesting as an intraventricular mass has been reported in the literature (6). Herein, we report a case of CNS-MM in a patient with 17p13 deletion (del 17p) manifested as an intraventricular mass with leptomeningeal involvement combined with perineural spread. This is the first reported case of a CNS-MM featuring a contemporaneous intraventricular mass and leptomeningeal involvement; both are very rare manifestations of CNS-MM. We also describe the unusual computed tomography (CT) and magnetic resonance imaging (MRI) findings. Central Nervous System Involvement in a Patient with Multiple Myeloma Manifesting as an Intraventricular Mass with Leptomeningeal Spread