Abstract
Esophageal duplication cyst (EDC) is a congenital malformation of the posterior primitive foregut, which mainly occurs in the thoracic esophagus. Here, we describe a 3-year-old Han Chinese boy afflicted with intermittent fever of acute onset and dry cough. Thoracic computed tomography revealed an 10 cm × 5.4 cm × 5.8 cm oval-shaped, cyst-like tumor located in the extrapleural space, extending along the right paravertebral gutter and compressing the trachea forward. An additional small-sized, oval-shaped cyst was identified in the posterior mediastinum, between the esophagus and the spinal column, at the T1 level. During open thoracotomy, under general anesthesia, an opaque, thick-walled, esophageal cyst was revealed not to be in communication with the esophageal lumen or the trachea. This cyst was subsequently resected in an en bloc manner. The small (1-cm) esophageal cyst was left untreated based on a “wait-and-see” policy. Histological analysis showed that the resected cyst was walled by hyperplastic, fibrous tissues and locally lined with gastric mucosa inherent glands. This finding was consistent with a diagnosis of EDC, with ectopic gastric mucosa. The respiratory tract symptoms resolved immediately after the operation. Computed tomography obtained at the 6-month follow-up showed that no disease, residual or recurrence, was present after the resection of the large-sized cyst, and the small-sized cyst remained unchanged in size.
Highlights
Gastrointestinal (GI) tract duplication, or GI duplication cyst, is a rare congenital malformation of the GI tract
We described a boy afflicted with intermittent fever of acute onset and dry cough, and diagnosed as having double Esophageal duplication cyst (EDC) containing ectopic gastric mucosa
One case of EDC with ectopic gastric mucosa in an infant presenting with hemoptysis has been reported [14]; respiratory tract bleeding in this patient may have resulted involvement [13]
Summary
Gastrointestinal (GI) tract duplication, or GI duplication cyst, is a rare congenital malformation of the GI tract. We described a boy afflicted with intermittent fever of acute onset and dry cough, and diagnosed as having double EDCs containing ectopic gastric mucosa. The smaller cyst was located between the posterior wall of the lower cervical esophagus, and the spinal column (level T1), at a size of 1.0 cm × 0.8 cm (Figure 1C). The small esophageal cyst was left untreated based on a “wait-and-see” policy This cyst was located at the thoracic entrance (the junction between the cervical portion and the thoracic portion of the esophagus) and was not accessible through the sixth intercostal space incision. Histological analysis of the resected esophageal cyst revealed that the cyst wall was composed of hyperplastic fibers and lined with stratified squamous epithelium, containing gastric mucosal simple columnar epithelium (Figure 4). A follow-up chest CT scan showed that no disease, residual or recurrent, was present after the resection of the large-sized cyst, and the small-sized cyst remained unchanged in dimension (Figure 5)
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