Background: To add to the confusion in the debate on pathogenesis of os-odontoideum, an additional report of its occurrence in two members of the same family is reported.
Case description: This is a report of a rare coincidence wherein both father (51 years old) and daughter (25 years old) were diagnosed to have os odontoideum and presented with crippling neurologic deficits.
Conclusions: Following atlantoaxial fixation, both recovered in their symptoms. Investigations revealed no genetic abnormality. Literature on the subject is briefly reviewed.
Keywords: Atlantoaxial fixation; Atlantoaxial instability; Os odontoideum.
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