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Background: Primary osteosarcoma of craniovertebral junction is extremely rare and reports in paediatric patients are limited. The symptoms are subtle and mostly underestimated.Case presentation.We present an unusual case of a 11-years old girl affected by an extensive and metastatic osteosarcoma of the atlas presented with cervicalgia and right torticollis refractory to medical treatment. The patient underwent open biopsy confirming the malignant histology. Conclusions: Cervicalgia with or without a torticollis refractory to medical treatment, in absence of history of fell or trauma, is highly suspicious and should be considered as a warning sign for a severe pathology and it should not be neglected.