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BACKGROUND: Cervicothoracic vertebral subluxation in sheep presents as a postural and locomotor disorder, and has been described in several breeds in Australia and overseas. Cervical myopathy may also be present in these cases. CASE REPORT: A New South Wales sheep producer reported a postural and locomotor disorder with a low prevalence in his Poll Merino stud flock, affecting neonate, weaner and adult sheep. Animals with postural abnormalities, variable degrees of ataxia and proprioceptive deficits involving both fore and hind limbs were described. Abnormalities of the cervicothoracic vertebral column were identified grossly during necropsy, with misalignment and consequent narrowing of the posterior cervical spinal canal. Lesions ranging from pallor (cellular degeneration) to white streaky lesions with pinpoint haemorrhage (necrosis) were identified in the cervicothoracic paravertebral musculature of affected animals. Boney abnormalities were further characterised by imaging studies. Pedigree analysis of the very extensive breeding and disease incident records available for this flock suggested that the disease was inherited. A similar case recognised in a separate New South Wales Poll Merino flock is also described. CONCLUSION: This report describes an entity of cervicothoracic vertebral subluxation in two Poll Merino sheep flocks, with cervical myopathy also identified in one, with preliminary evidence in the primary case that there is likely to be a hereditary basis. The two cases outlined in this report resemble the findings of several historical investigations into ovine flock postural disorders in Australia and beyond.