Background <p>Scoliosis is a common spine deformity. Most cases are idiopathic. However, it can be predisposed by certain congenital and neuromuscular conditions, such as osteogenesis imperfecta (OI). Surgical site infection (SSI) is a possible complication of scoliosis surgery. <i>Corynebacterium jeikeium</i> (<i>C. jeikeium</i>) is a rod-shaped, Gram-positive bacterium. It is part of the normal skin flora and has been implicated in various infections, especially in the immunocompromised. To date, only one case of SSI following scoliosis surgery has been reported to be secondary to C. jeikeium. In this report, we present a patient with OI who developed delayed SSI following scoliosis surgery secondary to <i>C. jeikeium</i>.</p> Case presentation <p>A 16-year-old male patient with OI underwent corrective surgery for his thoracolumbar scoliosis. Five months later, he presented with surgical site swelling with associated fever and pain following minor blunt trauma to his back. Further work-up confirmed the presence of deep SSI, and pus culturing yielded multidrug-resistant <i>C. jeikeium</i>. He was initially treated with surgical wound debridement and antibiotic therapy. However, the infection persisted, and he was managed with implant removal followed by a prolonged course of antibiotic therapy.</p> Conclusions <p>Our case represents the second reported case of SSI secondary to C. jeikeium following scoliosis surgery. C. jeikeium is a rare cause of SSI in the immunocompetent. It can cause indolent, yet persistent infections. Also, this case discusses how minor trauma could have led to this complication in an OI patient.</p>