This systematic review evaluates the strength of the evidence for the role of cervical musculoskeletal dysfunction in migraine. In this review, cervical musculoskeletal dysfunction will refer to the abnormal sensory afferentation from cervical region structures contained within the receptive field of the trigeminocervical nucleus. Electronic database searches using MEDLINE, PubMed and CINAHL were performed, and 17 studies investigating cervical musculoskeletal dysfunction in people with migraine were selected for review. The methodological quality of the included studies was assessed by two independent reviewers using a customized checklist. The review found that intersubject differences were inadequately reported and controlled, which resulted in grouping of participants with varying pathologies and symptoms. A diverse range of assessment procedures was used by the reviewed studies, which made comparison of their findings difficult. The assessment procedures were mainly used to quantify the degree of cervical musculoskeletal dysfunction, rather than to identify a cause and effect relationship between cervical structure and migrainous pain. Although animal study evidence proposes a role for cervical musculoskeletal dysfunction in migraine, this systematic review of the literature found that there is currently no convincing evidence to confirm this phenomenon in humans.