A case of cervical esophageal duplication cyst in a newborn infant.

Shoko Kawashima,Mariko Fujibayashi,Shuri Kimura,Yoshihiko Naritaka,Osamu Segawa,Nobuhide Henmi,Masayoshi Tsuchiya,Hisaya Hasegawa

doi:10.1186/s40792-016-0157-2

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.

Highlights

Esophageal duplication cysts are rare congenital cystic masses resulting from an error in foregut budding in the developing embryo and with a reported incidence of 1 in 8200 autopsies [1]
We report the case of a neonate with cervical esophageal duplication cyst at birth and describe the features of infantile cervical esophageal duplication cysts along with 19 infantile case reports
Foregut budding error may lead to bronchogenic cyst, esophageal duplication cyst, or “bronchopulmonary foregut malformations” (BPFM), a term proposed in 1968 by Gerle et al [5] to encompass the full spectrum of developmental aberrations of the embryonic foregut

Summary

Background

Esophageal duplication cysts are rare congenital cystic masses resulting from an error in foregut budding in the developing embryo and with a reported incidence of 1 in 8200 autopsies [1]. Case presentation A full-term newborn girl was transferred and admitted to our hospital because of swelling of the right anterior neck since birth (Fig. 1a). She did not manifest with respiratory distress and feeding difficulty on admission. Cervical ultrasonography (Fig. 1b) showed a simple cyst measuring 40 × 24 × 33 mm on the right neck. Incision of the muscle layer was made slightly lateral to the border of the cyst and esophagus to avoid injury of the esophageal mucosa. Pathologic examination of the cyst showed an internal layer with pseudostratified ciliated epithelium (Fig. 3a) with covering layers of smooth muscle and striated muscle (Fig. 3b). The patient has been doing well for 4 years

Findings

Discussion

Conclusions