A case of cervical esophageal duplication cyst in a newborn infant

Abstract

Esophageal duplication cyst is a rare congenital anomaly resulting from a foregut budding error during the fourth to sixth week of embryonic development. Cervical esophageal duplication cysts are very rare and may cause respiratory distress in infancy. A full-term newborn girl who was born by normal delivery was transferred to our hospital because of swelling of the right anterior neck since birth. Cervical ultrasonography showed a 40 × 24 × 33 mm simple cyst on the right neck. Tracheal intubation was required at 2 weeks of age because of worsening external compression of the trachea. Fine-needle aspiration cytology revealed the existence of ciliated epithelium. At 1 month of age, exploration was performed through a transverse neck incision. The cyst had a layer of muscle connected to the lateral wall of the esophagus. Histopathological diagnosis was a cervical esophageal duplication cyst. We describe the clinical features of infantile cervical esophageal duplication cysts based on our experience of this rare disease in a neonate, along with a review of 19 cases previously reported in literature.

Keywords: Cervical; Congenital duplication cyst; Esophagus; Newborn; Simple cyst.

Fig. 1

Clinical findings in a newborn infant girl with esophageal duplication cyst. a On physical examination, there was anterior neck swelling on the right. b Ultrasonography showed a simple cyst measuring 40 × 24 × 33 mm in the right neck. c T2-weighted enhanced magnetic resonance imaging revealed a simple cyst (asterisk) that displaced the sternocleidomastoid muscle anteriorly, the carotid sheath (arrow) to the right, and the larynx and trachea (arrow head) to the left. d Bronchoscopy showed compressive deformation of the trachea from the subglottis to the carina

Fig. 2

Intraoperative findings in an infant girl who underwent surgical exploration of an esophageal duplication cyst at 1 month of age. The cyst was separated from the right carotid artery and vein, thymus, vertebrae, and trachea. The cyst (asterisk) had a layer of muscle and was connected with the lateral wall of the esophagus (arrow head)

Fig. 3

Photomicrographs of a surgically resected infantile esophageal duplication cyst. a Pathologic examination showed an internal layer with pseudostratified ciliated epithelium (hematoxylin and eosin stain ×400) with b covering layers of smooth muscle (star) and striated muscle (arrow head) (hematoxylin and eosin stain ×40)