A 64-year-old female underwent salvage chemotherapy followed by allogeneic hematopoietic stem cell transplantation (alloHSCT). At 18 months after alloHSCT, she developed dysarthria and gait disturbances. Brain MRI revealed multifocal lesions with enhancement (Fig. 1A-D). A brain biopsy near the right callosal splenium showed patchy demyelination and T-cell infiltration (Fig. 1I-L). Spine MRI revealed multiple short-segment myelitis with enhancement (Fig. 1E-H). Since central nervous system (CNS) graft-versus-host disease (GVHD) after alloHSCT can lead to severe neurological complications, further research is needed to elucidate the risk factors of CNS GVHD and to manage it appropriately.1,2