Abstract
Atlanto-axial rotatory subluxation is a rare condition in childhood. A sudden onset of pain and limitation in neck movements are the most common presenting features. Usually, a previous trauma history exists. This study presents a case of an 18-month-old male with neck stiffness and pain in neck through palpation without trauma in his history but with rotatory subluxation of 30° in atlanto-axial joint observed in his cervical imaging. With this case, the aim was to emphasize the necessity for considering the atlanto-axial subluxation in patients less than five years old diagnosed with neck stiffness in differential diagnosis even if a trauma is not available in their histories.