A 14 year-old girl with a history of idiopathic scoliosis underwent surgical correction of scoliotic deformity with posterior spinal instrumentation and fusion using a spinal implant containing titanium alloy (T1-641-4VASTMF-130). The postoperative period was uncomplicated in general; except for a wound dehiscence at the fourth day post surgery that was repaired surgically with debridement and immediate skin closure. During the scheduled follow-up visits at 1st and 3rd month the clinical and radiographic image of the patient was normal. Surgical wound was completely healed with no signs of inflammation. Correction of scoliotic deformity both in frontal and sagittal planes has been achieved and patient was satisfied with overall outcome. Five months postoperatively, the parents seeked pediatric consultation because their child developed macroscopic hemuresis (blood in urine) and diffuse erythema. The patient was in good general condition. Laboratory studies on admission revealed that the white blood cell count was 9100/mL; granulocytes, 39%; monocytes, 6%; lymphocytes, 19%; and eosinophils, 36%. The hematocrit was 35.5% and the platelet count was 185 × 103/mL. C-reactive protein (CRP) was 5.0 mg/dL (0.7 to 1.7 mg/dL). Erythrocyte Sedimentation Rate was 75 mm. Modest elevations in transaminases and bilirubin were noted: total bilirubin was 1.4/dL, AST 59 and ALT 34. Renal biochemistry showed no abnormalities: blood urea nitrogen was 11 mg/dL and creatinine 0.8 mg/dL. Urine trace was positive for bilirubin, ketones, and protein. The urinary sediment contained 8 RBCs per high-power field and 7 WBCs per high-power field. No cellular casts were identified. Complement levels were normal. Antinuclear antibodies, rheumatoid factor, anti-neutrophil cytoplasmic antibodies, antibodies to glomerular basement membrane were normal. Hepatitis B surface antigen, hepatitis C antibody, and HIV antibodies were negative. Differential diagnosis included late periprosthetic infection, low virulence viral or bacterial infection unrelated to surgery, allergic reaction to metal implants or to other envinmental agents, and toxicity. Skin patch testing for metal hypersensitivity was strongly positive for titanium and nickel, supporting the role of the titanium implants in the development of secondary systemic vasculitis. The patient received corticosteroids systematically (hydrocortisone 10 mg) for 6 months, leading to total recess of erythema, hemuresis and proteinuria. Orthopedic surgery was consulted to consider removal of the spinal implants. After weighing the risks and benefits of the procedure, the titanium prosthesis was not removed, because spinal fusion was premature and early removal of instrumentation could inevitably lead to loss of reduction. However, 1 month post corticosteroid cessation a palpable mass close to surgical wound and a small skin dehiscence of the surgical scar was developed. A soft tissue ultra-sonography showed the presence of cystic formation 3 × 6 cm within the muscle layers of the thoraco-lumbar region and close to the spinal implants. Surgical debridement was decided which revealed the presence of pus with gram positive staining. There was some callus formation over the decorticated and grafted posterior elements as well as the osteotomized facet joints which was proved soft when we tried to remove the cross-links. Notwithstanding there was some minimal "elastic" motion of the spine after removing the metalwork. Therefore, one stage revision of posterior spinal instrumentation was performed as pus collection was deep and very close to the spinal implants. Although we were prepared for revision of the instrumentation with titanium and nickel-free implants, the presence of pus in the surgical field made evident that the etiology of vasculitis was a late infection and not a metal allergy. Therefore, we proceeded to a meticulous surgical debridement and re-implantation of titanium spinal implants which are associated with decreased rates of infection comparing to stainless steel. Intraoperative sample cultures were positive to Staphylococcus aureus. Intravenous antibiotics (were administered for three weeks followed another 3 week period of oral administration. The postoperative recovery was uneventful and the patient had complete resolution of symptoms. Neurologic examination was normal at the 6-week postoperative visit and the x-ray imaging in postero-anterior and lateral views showed no loss of initial reduction or implant loosening. Twenty-four post revision surgery, the patient is now free of symptoms without any signs of recurrence of either allergy or infection. There is no need for pain medication and the patient is back to daily activities without restrictions.