Hirayama disease, also known as monomelic amyotrophy, is a relatively benign motor neuron disease, characterized by insidious muscle wasting and weakness, unilateral or asymmetric restricted to the distal upper limb, followed by a quiescent phase after onset in the second to third decades.1 Pathophysiology is uncertain and no treatment is available to restore weakness.2 Although the degree of disability does not interfere with activities of daily living, 2,3 the appearance of the hand is of major concern to the patient but is often minimized by clinicians concerned about the cause and the weakness. Here, we describe hand rejuvenation surgery, a procedure that restores youthful appearance to the hand, as a treatment of a patient's chief complaint in Hirayama disease.Level of evidence is class IV: single observational study without controls.Case report. Hirayama disease was diagnosed in a 36-year-old man with chief complaints of left hand atrophy and weakness which was noted soon after age 13 and did not progress after age 20. On examination, marked atrophy of left intrinsic hand muscles and forearm flexors was noted. Slight weakness was demonstrated in the C7, C8, and T1 myotomes. EMG disclosed a slightly active and chronic neurogenic pattern in the appropriate spinal segments. MRI revealed asymmetric anterior flattening and atrophy of midlower cervical cord. He consulted several neurologists who recommended occupational therapy with no benefit. Because of his great concern about hand appearance, "rejuvenation" hand surgery was recommended by a plastic surgeon.Hand rejuvenation procedure. This is a combination of 2 procedures. In the first procedure, infraumbilical fat was harvested using a liposuction cannula. The fat was then washed and refined. A total of 20 mL of fat parcels were serially injected into the first through fourth interosseous subcutaneous spaces with multiple passes at 0.25 mL increments. Fat molding was performed by gentle digital manipulation to achieve a uniform distribution. After structural fat grafting, the patient returned for moulage, the process of forming the shape and volume of the prosthesis using dental alginate to generate the permanent prosthesis out of the silicone rubber. In the second procedure, an incision was made through the skin and subcutaneous fat, the fascia was opened and separated from the muscle belly, which was severely atrophic. Soft silicone fillers were placed into anatomic positions of the first and fifth interosseous fascial compartments. Pressure dressing was applied after the skin was closed. The patient tolerated the procedures well. Tissue edema was the only postoperative sequela but gradually resolved. The patient immediately recognized a major improvement of his hand appearance (figure). Gradual loss of hand fullness secondary to partial graft resorption is noted 5 years after the procedures; nevertheless, the patient stated that he would have had this procedure performed again.