Bilateral neonatal submandibular cystic hygroma with critical airway compromise: a stepwise multimodal management approach in a resource-limited setting: a case report

Abstract

Background: Cystic hygroma is a congenital lymphatic malformation most commonly involving the cervical region. Bilateral submandibular involvement in neonates is extremely rare and may rapidly progress to life-threatening airway obstruction due to proximity to the upper airway and the limited neonatal physiological reserve. Case presentation: We report a 29-day-old female neonate presenting with progressively enlarging bilateral submandibular cystic swellings associated with feeding difficulties and respiratory distress, necessitating nasogastric feeding. On examination, the infant had significant airway compromise requiring intubation. Imaging confirmed multiloculated cystic lesions causing marked compression of the upper airway without vascular invasion. Initial management involved endotracheal intubation and ultrasound-guided aspiration, leading to transient improvement. Intralesional doxycycline sclerotherapy was subsequently performed, resulting in partial regression and successful extubation. However, recurrence of respiratory distress necessitated reintubation and definitive surgical excision. Intraoperatively, prior sclerotherapy induced fibrosis, facilitating safer dissection with reduced bleeding. The postoperative course was uneventful, with full recovery. Discussion: This case demonstrates the importance of a structured stepwise approach to neonatal lymphatic malformations. While aspiration and sclerotherapy provide temporary stabilization, definitive surgical excision remains necessary in complex multiloculated lesions. Intralesional doxycycline may offer dual benefits by reducing cyst volume and improving surgical planes through fibrosis, supporting its adjunctive role in resource-limited settings. Conclusion: Early airway stabilization followed by staged sclerotherapy and timely surgical intervention is essential for optimal outcomes in neonatal cystic hygroma.