Studies in normal volunteers have demonstrated that the lateral atlanto-axial joints (C1-2) are capable of causing pain in the occiput, but few clinical studies have validated this source of occipital headache. The present study tested the null hypothesis that the lateral atlanto-axial joints are not a common source of occipital headache. Patients presenting with occipital pain underwent diagnostic blocks of their lateral atlanto-axial joints if they demonstrated clinical features presumptively suggestive of a C1-2 origin for their pain. Of 34 patients investigated, 21 obtained complete relief of their headache following diagnostic blocks, indicating that a C1-2 source of occipital pain is not rare. The clinical features used to select patients for blocks, however, had a positive predictive value of only 60%. Further study of headaches from C1-2 seems justified in order to establish more definitively the prevalence of this condition and how it might become better recognized in practice.