A seven-month-old infant was admitted to our hospital with a 1-wk history of shortness of breath, dysphagia, and fever. Diagnosis of esophageal perforation following fish vertebra ingestion was made by history review, pneumomediastinum and an irregular hyperdense lesion noted in initial chest radiogram. Neck computed tomography (CT) confirmed that the foreign body located at the cricopharyngeal level and a small esophageal tracheal fistula was shown by esophagogram. The initial response to treatment of fish bone removal guided by panendoscopy and antibiotics administration was poor since pneumothorax plus empyema developed. Fortunately, the patient's condition finally improved after decortication, mediastinotomy and perforated esophagus repair. To our knowledge, this is the first case report of esophageal perforation due to fish bone ingestion in infancy. In addition to particular caution that has to be taken when feeding the innocent, young victim, it may indicate the importance of surgical intervention for complicated esophageal perforation in infancy.