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Upper limb length discrepancy is a rare occurrence.Humerus shortening may need specialized treatment to restore the functional and cosmetic status of upper limb.We report a case of humerus lengthening of 9 cm with a monorail extal fixator and the result was observed during a 2-year follow-up.Humerus lengthening needs specialized focus as it is not only a cosmetic issue but also a functional demand.The monorail unilateral fixator is more functional and cosmetically acceptable,and thus becomes an effective treatment option.