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Large lingual heterotopic gastrointestinal cyst in a newborn: A case report
Atsushi-Doksa Lee1, Kazuma Harada1, Susumu Tanaka1
1First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Suita 565-0871, Osaka, Japan.
Insights
A rare congenital tongue cyst in a newborn was successfully treated with aspiration and surgical resection. This intervention resolved airway distress and feeding difficulties, ensuring a positive outcome.
Area of Science:
- Pediatric Surgery
- Congenital Abnormalities
- Gastrointestinal Pathology
Background:
- Heterotopic gastrointestinal cysts are rare, characterized by gastrointestinal epithelium in the cyst wall.
- Oral cavity presentation, particularly in neonates, is uncommon.
- Congenital tongue cysts diagnosed via ultrasonography are exceptionally rare.
Observation:
- A 12-day-old female infant presented with anterior tongue swelling, noted antenatally via ultrasonography.
- The infant experienced dyspnea shortly after birth, necessitating cyst aspiration.
- Post-aspiration, the infant's breathing improved, enabling breastfeeding.
Findings:
- Histopathologic examination confirmed the cyst wall comprised gastrointestinal-type columnar and pseudostratified ciliated epithelium.
- Surgical excision was performed on day 67 under general anesthesia.
- The patient resumed breastfeeding 3 hours post-surgery with an uneventful recovery.
Implications:
- Cyst aspiration effectively managed acute airway distress and feeding issues.
- Surgical resection provided a definitive treatment with no perioperative complications.
- This case highlights a successful multidisciplinary approach to a rare congenital anomaly.
Background:
Heterotopic gastrointestinal cysts have gastrointestinal epithelium in the cyst wall and rarely occur in the oral cavity. Most are found in the neonatal period. However, heterotopic gastrointestinal cysts that are diagnosed as a congenital tongue cyst by routine ultrasonography are extremely rare.
Case Summary:
A 12-day-old female presented with swelling of the anterior tongue. The obstetrician had detected significant tongue swelling on fetal ultrasonography in the 35th gestational week. The female was born by cesarean delivery at gestational week 39. She soon became dyspneic, and the cyst was aspirated. After the aspiration, her breathing recovered and she started breastfeeding. The cyst was excised under general anesthesia on the 67th day. Histopathologic examination showed that that cyst wall consisted of a lining of columnar gastrointestinal-type epithelium and pseudostratified ciliated epithelium. The patient restarted breastfeeding 3 h after surgery. The postoperative course was uneventful.
Conclusion:
Airway distress and feeding difficulty were successfully avoided by cyst aspiration, and surgical resection was performed with no perioperative complications.
