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Evidence suggests that elevated thyroid function and elevated levels of thyroid autoantibodies are associated with risk of moyamoya disease (MMD). Therefore a meta-analysis of all available evidence was performed, including unpublished data from our own center, in order to assess this association.Major literature databases were reviewed without language restrictions to identify studies examining the association between MMD and thyroid function or thyroid autoantibodies. These data were combined with those from our own prospective study. This study involved consecutively recruited patients with MMD and two groups of age-matched control patients: patients with non-MMD stroke and healthy individuals. The groups were compared in terms of serum triiodothyronine (T3), thyroxine (T4), free T4, free T3, thyroid stimulating hormone, anti-thyroperoxidase antibody and anti-thyroglobulin antibody.Our prospective study involved 28 patients with MMD, 28 age-matched patients with non-MMD stroke and 28 age-matched healthy control participants. The results showed an association between risk of MMD and elevated thyroid autoantibodies [odds ratio (OR) 9.00, 95% confidence interval (CI) 1.03–78.94] but not between risk of MMD and elevated thyroid function (OR 5.87, 95% CI 0.64–53.93). Meta-analysis of our data with findings from the literature further supported the association with elevated thyroid autoantibodies (OR 8.77, 95% CI 4.45–17.29) and indicated an association with elevated thyroid function (OR 9.74, 95% CI 2.18–43.49).Evidence strongly suggests that elevated thyroid autoantibodies and elevated thyroid function are independently associated with MMD. These clinical variables may require regular monitoring in patients with MMD.