Ganglia in soft tissue are common and usually occur in close relations with joints, tendon sheaths, or tendons. However, intra-osseous ganglia are very rare. We report a case of intra-osseous ganglion arising from the right sided proximal humerus in a 33 year-old woman. It was multiple cysts surrounded by thin rims of sclerotic bone in the subchondral epiphysis without other features of degenerative joint disease or destruction of adjacent soft tissue.