-Report on Experiments and Clinical Cases-

Fatal Tracheo-innominate Artery Fistula after Tracheostomy in a Patient with Pelizaeus-Merzbacher Disease

Takehide Imai¹, Masato Takase¹, Sachiyo Takeda¹, Katsuji Hosone², Shunichi Tomiyama³ and Yuichi Nakanowatari⁴

¹Department of Pediatrics, Nippon Medical School Tama Nagayama Hospital
²Department of Pathophysiology, Nippon Medical School Tama Nagayama Hospital
³Department of Otolaryngology, Nippon Medical School Tama Nagayama Hospital
⁴Department of Emergency and Critical Care Center, Nippon Medical School Tama Nagayama Hospital

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.

J Nippon Med Sch 2012; 79: 274-279

Keywords
tracheo-innominate artery fistula, tracheostomy, complication, Pelizaeus-Merzbacher disease, noninvasive ventilation

Correspondence to
Takehide Imai, Department of Pediatrics, Nippon Medical School Tama Nagayama Hospital, 1-7-1 Nagayama, Tama, Tokyo 206-8512, Japan
takehide@nms.ac.jp

Received, September 8, 2011
Accepted, December 8, 2011