| Summary: | Background: Chondromyxoid fibroma (CMF) is a very rare case usually appears around the knee. CMF presented high recurrence rate 25% among treated cases. However, in this case, interestingly CMF in the right upper arm was radiologically and pathologically diagnosed. It measures a 6.9 mm × 1.7 mm lesion. Case presentation: 36-year-old male showed CMF in the juxtacortic proximal humerus. Computed tomography (CT) and magnetic resonance imaging (MRI) of the right upper arm interestingly presented an expansile, mildly lobulated, septate lesion a 6.9 mm × 1.7 mm. Pathological analysis of biopsy showed a bony tissue consisting of cartilaginous, fibrous and myxoid stroma with abundant stellate shaped cells that present vary nucleus size and a minority of nuclear division. Surgical allogeneic bone graft and plate internal fixation was performed after the intralesional curettage surgery. Postoperation the patient was received doxorubicin 50–75 mg/m2 21 days cycle. Results showed that the intralesional curettage surgery of CMF, allogeneic bone graft and plate internal fixation combined with doxorubicin was successfully treated CMF and efficiently prevent relapse during 20 months of observations. Conclusion: the juxtacortic CMF was unusually diagnosed in the proximal humerus, it has been efficiently eliminated by a surgical operation and doxorubicin 50–75 mg/m2 that prevented tumor recurrence. Keywords: Chondromyxoid fibroma, Juxtacortic, Proximal humerus, Bone fibroma
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