title: Fatal tracheo-innominate artery fistula after tracheostomy in a patient with Pelizaeus-Merzbacher disease.
abstract: Tracheo-innominate artery fistula (TIF) is a serious, life-threatening complication following tracheostomy. We report a fatal TIF in a 15-year-old girl with Pelizaeus-Merzbacher disease. She received a tracheostomy for prolonged translaryngeal intubation due to acute respiratory failure without a trial of noninvasive ventilatory support before intubation. Severe hemorrhage from the TIF occurred 6 months after tracheostomy; immediate resuscitation failed. Antemortem fiberoptic bronchoscopy showed tracheal stenosis accompanied by granulation tissue, and postmortem examination revealed TIF with ulcerative granulation. Preventive intervention is required to avoid catastrophic TIF due to its high mortality rate. Moreover, to avoid prolonged translaryngeal intubation leading to tracheostomy, noninvasive ventilatory support before translaryngeal intubation, if applicable, is beneficial.
authors.affiliation: Department of Pediatrics, Nippon Medical School Tama Nagayama Hospital, Tama, Tokyo, Japan. takehide@nms.ac.jp
authors.forename: Takehide
authors.initials: T
authors.lastname: Imai
authors.forename: Masato
authors.initials: M
authors.lastname: Takase
authors.forename: Sachiyo
authors.initials: S
authors.lastname: Takeda
authors.forename: Katsuji
authors.initials: K
authors.lastname: Hosone
authors.forename: Shunichi
authors.initials: S
authors.lastname: Tomiyama
authors.forename: Yuichi
authors.initials: Y
authors.lastname: Nakanowatari
mesh_terms: D000293:Adolescent; D016122:Brachiocephalic Trunk / Q000000981:diagnostic imaging / Q000601:surgery*; D001999:Bronchoscopy; D017809:Fatal Outcome; D005260:Female; D006097:Granulation Tissue / Q000473:pathology; D006801:Humans; D007223:Infant; D020371:Pelizaeus-Merzbacher Disease / Q000150:complications* / Q000000981:diagnostic imaging / Q000601:surgery; D013902:Radiography, Thoracic; D014057:Tomography, X-Ray Computed; D014132:Trachea / Q000098:blood supply* / Q000000981:diagnostic imaging / Q000473:pathology / Q000601:surgery*; D014139:Tracheostomy / Q000009:adverse effects*; D016157:Vascular Fistula / Q000000981:diagnostic imaging / Q000209:etiology*