Our study suggests that coexisting celiac sprue is one of the mechanisms responsible for the malabsorption associated with idiopathic hypoparathyroidism. Celiac sprue is relatively common, and this association could occur by coincidence. Alternately there may be shared determinants of susceptibility or perhaps a direct immunological relationship. In such patients, both gluten-free diet and correction of the hypoparathyroid state may be necessary to correct malabsorption. In our literature survey, this is the first case in which celiac sprue associated with idiopathic hypoparathyroidism was documented. The importance of intestinal biopsy in evaluating malabsorption in patients with idiopathic hypoparathyroidism is emphasized.