Abstract: Multicentric Castleman disease (MCD), a rare lymphoproliferative disorder of incompletely understood etiology, is manifested as enlarged multiple lymph node and multiple organs involvement. The best therapeutic option for this disease remains unknown. A 21-year-old male patient of MCD with swilling lymph nodes received combination chemotherapy treatment with unsatisfactory efficacy. He developed thrombocytopenia during disease progression and failed steroid treatment. His platelet increased to normal level on day 27 of receiving infusion of humanised anti-CD19 chimeric antigen receptor (CAR) T cells (1 × 10⁶ cells/kg) and murine anti-BCMA CAR T cells (0.6 × 10⁶ cells/kg). He has a stable platelet count and no evidence of progression of lymphadenopathy for 45 months at this writing. This case report reminds clinicians to be aware of the potential clinical value of CAR T cells therapy in MCD.