Author(s)
Josiah P Brandt, MD
James B Tansey
Affiliation(s)
University of Tennessee Health Science Center;
Abstract:
Objective: Infantile hemangiomas (IHs) are common pediatric tumors. IHs located in the airway can cause stridor and a barking cough. Standard therapy involves propranolol, but occasionally surgical intervention is required if lesions persist. The purpose of this study is to examine the case of an infant with an airway IH which was unresponsive to propranolol and developed into a mucus-filled cystic structure requiring surgical excision via laryngofissure.
Method: A 2-month-old male patient with a history of stridor was admitted for increased respiratory distress. Direct laryngoscopy and rigid bronchoscopy (DLB) showed 75% airway occlusion with uncertain etiology. Intubation and later a tracheostomy were required for survival. At 6 months of age, DLB revealed a subglottic IH causing 90% airway occlusion. Thus, propranolol was prescribed. At 13 months of age, the IH appeared unchanged, so excision via an anterior laryngofissure was performed. Visualization of the subglottic mass revealed a thick-walled cystic structure. The cystic sac was confirmed via DLB to be the mass that had been diagnosed as an IH. This mass was successfully removed, revealing a nearly patent subglottis. GLUT1 staining of the specimen confirmed the diagnosis of hemangioma.
Results: The patient is doing well with a patent glottis and subglottis. He is currently undergoing institutionalized decannulation protocol.
Conclusion: This case report demonstrates an unusual disease course for an airway IH. No similar cases of an airway IH developing into a mucus-filled, cystic structure are found in the literature. Furthermore, IHs requiring laryngofissure are rare and interesting from a surgical perspective.