Asphyxiation due to obstructive fibrinous tracheal pseudomembrane after closure of repeated tracheostomy in a case of von Recklinghausen disease

冯·雷克林豪森病患者反复气管切开术后,因阻塞性纤维蛋白性气管假膜形成导致窒息。

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Abstract

BACKGROUND: Obstructive fibrinous tracheal pseudomembrane (OFTP) is a rare but potentially fatal complication, most commonly reported after endotracheal intubation. Its occurrence following tracheostomy closure is poorly recognized. CASE PRESENTATION: We report a fatal case of OFTP in a 33-year-old man with von Recklinghausen disease who developed progressive airway obstruction following closure of a repeated tracheostomy. The patient had a history of multiple facial tumor resections requiring repeated tracheostomies due to orofacial deformities. After the eighth tracheostomy and successful tumor debulking, the tracheal cannula was removed on postoperative day 6. At that time, CT revealed only mild tracheal narrowing without intraluminal obstruction, and the patient remained asymptomatic. However, he was found in cardiopulmonary arrest on postoperative day 14. Postmortem CT showed near-complete tracheal obstruction and pulmonary edema. Autopsy confirmed a grayish-white pseudomembrane obstructing the tracheal lumen, with only a 2-mm residual gap. Histopathology revealed fibrinous exudate with neutrophilic infiltration, consistent with OFTP. CONCLUSION: This case illustrates that OFTP can occur silently after tracheostomy closure and may lead to sudden death. Vigilant monitoring with CT or bronchoscopy should be considered in high-risk patients with repeated tracheal interventions.

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