We experienced a case of copper‐deficient anemia in a premature infant with intestinal atresia（IA）．He was born at 35 weeks’gestational age weighing 2,890 g. Jejunostomy was created at day 1 because of the IA. He received total parental nutrition and elemental diet postoperatively. Progressive anemia developed from postoperative day 9 without iron deficiency. He was diagnosed with copper‐deficient anemia because his serum ceruloplasmin concentration was low as 13.7 mg/dl while, in addition, his neutrophil count was found to have decreased. Copper supplementation was performed with a copper‐rich enteral supplement. The serum concentration of copper and ceruloplasmin immediately improved. At day 57, the ileostomy was closed, and complete breast‐feeding was started after the operation. He was discharged at day 73 without progression of anemia. Our patient’s copper deficiency might have been due to insufficient copper accumulation caused by his preterm birth and impaired copper absorption via the jejunum. We must pay close attention to copper deficiency in preterm infants with enterostomy．

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