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In the last few years, several scientific papers and reports have demonstrated magnetic resonance (MR) signal intensity (SI) changes on pre-contrast T1-weighted images following multiple gadolinium-based contrast agents (GBCA) administrations, particularly following the exposure to linear GBCAs. Pathological animal and human post-mortem studies have confirmed the relationship between this radiological finding and the presence of gadolinium accumulation in vulnerable brain regions in patients with normal renal function. In this short communication, we report the case of a 15-year-old patient affected by b-cell acute lymphoblastic leukemia (bALL) who developed a hyperintense signal in the dentate nuclei following multiple administrations of a macrocyclic GBCA. The purpose of this report is to discuss possible differential diagnoses of this radiological finding with special focus on the differentiation between iron or manganese accumulation, post-irradiation changes and GBCA-related Gd deposition, highlighting the importance of the acquisition of accurate clinical data to improve our scientific knowledge.