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Restoration of neutral mechanical alignment of the lower limb is an important factor in the treatment of unicompartmental arthrosis. Traditionally, medial opening wedge high tibial osteotomy has been widely performed to correct varus malalignment with unicompartmental arthrosis. However, an ideal indication for the high tibial osteotomy is the knee with metaphyseal tibial varus malalignment. The basic principle of corrective osteotomy is performing an osteotomy at the center of the deformity to prevent abnormal joint line obliquity. If pathologic distal femoral varus deformity is the cause of genu varum, the osteotomy should be performed in the distal femur. Reports of medial opening wedge distal femoral osteotomy (DFO) to correct varus malalignment are rare. We present a case of this very rare and challenging condition in a 47-year-old male, which was successfully treated by medial opening wedge DFO.