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Background: Atlanto-axial rotatory fixation is a persistent deformity of the C1-2 vertebral relationship caused by subluxation of the articular surfaces, and can occur after positioning for ENT procedures where the head is rotated - for example to access the ear or posterior triangle of the neck. If promptly recognised, it can usually be managed successfully with conservative methods, without long-lasting sequelae, but delayed or inappropriate management may lead to permanent neck deformity, neurological problems and pain. Method: Case review. Case report: Two children with atlanto-axial rotatory fixation following ENT surgery; one child was referred early and managed successfully, and one had delayed referral resulting in permanent severe positional deformity. Conclusion: Atlanto-axial rotatory fixation is easily missed; there are significant clinical and medicolegal implications if it is not promptly recognised. A suggested management algorithm is presented. [ABSTRACT FROM AUTHOR]