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Published on: April 7, 2023
A case of cervical esophageal duplication cyst in a newborn infant
Shoko Kawashima1,2, Osamu Segawa3,4, Shuri Kimura2
1Department of Surgery, Tokyo Women's Medical University Medical Center East, 2-1-10 Nishi Ogu, Arakawa-ku, Tokyo, 116-8567, Japan.
Insights
Cervical esophageal duplication cysts are rare congenital anomalies causing neck swelling and respiratory distress in infants. Early diagnosis and surgical intervention are crucial for managing this condition.
Area of Science:
- Pediatric Surgery
- Congenital Anomalies
- Embryology
Background:
- Esophageal duplication cysts are rare congenital anomalies arising from embryonic foregut development errors.
- Cervical esophageal duplication cysts are exceptionally rare and can lead to significant respiratory distress in neonates.
- This condition presents as neck swelling and may compress the trachea, necessitating prompt medical attention.
Purpose of the Study:
- To describe the clinical presentation and management of a rare infantile cervical esophageal duplication cyst.
- To review existing literature on infantile cervical esophageal duplication cysts.
- To highlight the importance of early diagnosis and surgical intervention for this condition.
Main Methods:
- Case report of a neonate with a cervical esophageal duplication cyst.
- Diagnostic imaging including cervical ultrasonography.
- Fine-needle aspiration cytology and histopathological examination.
- Surgical exploration and cyst excision.
- Literature review of 19 previously reported cases.
Main Results:
- A neonate presented with right neck swelling and required tracheal intubation due to external compression.
- Ultrasonography identified a simple cyst; fine-needle aspiration revealed ciliated epithelium.
- Histopathology confirmed a cervical esophageal duplication cyst with muscular connection to the esophagus.
- Review of 19 cases provided insights into the clinical spectrum and management.
Conclusions:
- Cervical esophageal duplication cysts, though rare, pose a significant risk of respiratory compromise in infants.
- Multimodality diagnostic approaches are essential for accurate diagnosis.
- Surgical excision is the definitive treatment for symptomatic cysts.
- This case and literature review underscore the need for awareness and timely intervention in managing infantile cervical esophageal duplication cysts.
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.
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