We describe a case of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO syndrome) in an 8-year-old boy with an unusual presentation of acute transitory hemiparesis. SAPHO syndrome has been reported in association with inflammatory bowel diseases, chest complications, and pulmonary involvement. No patient with both SAPHO syndrome and neurologic complaints has been previously described. Further observations are needed to confirm if SAPHO syndrome and hemiparesis represent a coincidence or a new association.