{Reference Type}: Case Reports {Title}: Juvenile xanthogranuloma manifesting with LCH-associated neurodegenerative disease-like radiological findings. {Author}: Daifu T;Umeda K;Yokoyama A;Yoshida T;Saida S;Kato I;Hiramatsu H;Kudo K;Higuchi Y;Takita J; {Journal}: Pediatr Blood Cancer {Volume}: 71 {Issue}: 7 {Year}: 2024 Jul 28 {Factor}: 3.838 {DOI}: 10.1002/pbc.31043 {Abstract}: Here, we describe two patients with juvenile xanthogranuloma (JXG) manifesting with Langerhans cell histiocytosis (LCH)-associated neurodegenerative disease (ND)-like radiological findings. One patient showed typical radiological abnormalities at onset, which worsened with progressing central nervous system symptoms 7 years after LCH-oriented chemotherapy. Another showed spontaneous regression of clinical symptoms, with a transient radiological change 1 year after salvage chemotherapy for recurrence of JXG. These data regarding JXG-associated ND will facilitate future investigation of the disease, as well as development of therapeutic interventions.