Introduction: Atlanto-axial rotatory fixation (AARF) is a rare complication of ventriculo-peritoneal shunt (VPS) surgery.
Case presentation: The authors present a unique case of AARF developing early after VP shunting, with persistent torticollis, a "cock-robin" head position, and a thick fibrous band along the catheter path. Due to refractoriness to conservative treatments, AARF, which can be an early-onset complication of VPS surgery, was resolved by removing the distal catheter along with the fibrous band encasing it.
Conclusion: Surgical removal of the fibrous band might be enough to solve such complication with no need of further surgical fusion procedures.
Keywords: Atlanto-axial rotatory fixation; Hydrocephalus; Pediatrics; Ventriculo-peritoneal shunt.