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A 69-year-old woman diagnosed with Waldenström macroglobulinemia (WM) was referred for 18F-FDG and 68Ga-Pentixafor PET/CT for evaluation of the disease. Extensive involvement of WM with intense radioactivity in the bone marrow and lymph nodes was found in 68Ga-Pentixafor PET/CT. However, in 18F-FDG PET/CT, the lesions showed significantly lower tracer uptake. Given the superiority of 68Ga-Pentixafor in detecting WM lesions to 18F-FDG in this patient, 68Ga-Pentixafor PET/CT might be a promising imaging modality in assessment of tumor burden of WM in future studies.