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Abstract Background Isolated involvement of bone marrow in sarcoidosis has not been reported commonly. Sarcoidosis is a systemic granulomatous disease of unknown origin, characterized by the presence of non-caseating granulomatous lesions. There should be high index of suspicion in patients having underlying lymphoproliferative malignancies. Case presentation We present a 27-year-old male, known case of chronic myeloid leukemia, presenting as fever of unknown origin diagnosed with isolated bone marrow sarcoidosis.