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Copper deficiency is an uncommonly reported complication of celiac disease that has not received much attention in recent years. Copper deficiency may result in anemia and thrombocytopenia and also irreversible myeloneuropathy if it is not detected and treated appropriately. The prevalence of copper deficiency in patients with celiac disease is unknown. We describe 5 patients with celiac disease and associated copper deficiency diagnosed at our institution in recent years. All 5 patients had neurologic complications of copper deficiency and 3 patients also presented with hematologic abnormalities. We also review the literature regarding copper deficiency in celiac disease.