We applied RNA-protein interactome capture method called XRNAX to shed light on RNA-bound proteome in the brain affected with dysmyelination. We found that sets of canonical RBPs that are known to regulate alternative splicing and being engaged in the formation of cytoplasmic granules are perturbed at the level of their RNA interactome in the mouse brain with the inborn deficit of myelin. This dataset contains the whole brain proteomics data as well as LC-MS/MS data from XRNAX extracts enriched in brain RBPs following UV crosslink of RNA-protein interaction in the brain tissue homogenates from wildtype C57BL6/N (experiments 1 and 2) and shiverer mouse model (experiment 3). Whole brain tissues were homogenized using two different methods: biopulverization of the flash-frozen tissue in liquid nitrogen (experiments 2 and 3), and alternatively, tissues submersed in PBS buffer immediately after dissections were disintegrated with Dounce homogenizer (experiment 1).