Anterior mediastinal tracheostomy for severe tracheal stenosis in a child with severe motor and intellectual disabilities: a case report.

BACKGROUND: Owing to recurrent aspiration pneumonia and airway stenosis secondary to thoracic deformities, tracheostomy or laryngotracheal separation are often necessary in children with severe motor and intellectual disabilities. However, these procedures are associated with the risks of tracheal stenosis due to tracheal granulation and tracheoinnominate artery fistula formation. We report a case of a child with severe motor and intellectual disabilities treated with an anterior mediastinal tracheostomy. CASE PRESENTATION: The patient was a 15-year-old boy with severe motor and intellectual disabilities due to intractable epilepsy. Due to thoracic deformity and tracheomalacia, the patient had a flattened and narrowed trachea. Accordingly, laryngotracheal separation was performed 4 months before admission to avoid aspiration pneumonia. Due to a common cold, the patient required frequent sputum suctioning, which exacerbated the tracheal stenosis. Bronchoscopy revealed tracheal stenosis 4-5 cm caudal to the tracheostomy site, tracheal mucosal ulcers, and pulsation of the innominate artery on the anterior wall of the trachea. We performed an anterior mediastinum tracheostomy to release the tracheal stenosis and prevent tracheoinnominate artery fistula formation. CONCLUSIONS: Anterior mediastinal tracheostomy has several advantages. Including sufficient release of bony compression, release of tracheal hyperextension, and relief of tracheal and innominate artery contact ensures a cannula-free tracheostomy, and there is no need to dissect the brachiocephalic artery. It is the procedure of choice in cases of head and neck malignancies requiring extensive tracheal resection and could be a good surgical option for severe tracheal stenosis and tracheoinnominate artery fistula in children with severe motor and intellectual disabilities.

Tracheoplasty for Tracheal Collapse after the Tracheoinnominate Fistula Repair / 日本心臓血管外科学会雑誌

A 13-year-old boy underwent tracheostomy due to post-cardiac arrest encephalopathy in our hospital. During the second postoperative month, massive bleeding from the tracheostomy tube lumen was observed; tracheoinnominate artery fistula was diagnosed. Two weeks postoperatively, the trachea collapsed; tracheoplasty with VA-ECMO was performed. The patient was placed in respiratory distress and the tracheostomy cannula was removed. The damaged part of the trachea was trimmed to form a fusiform structure, while the horizontal mattress suture technique was used for tracheoplasty. An endotracheal tube was then placed just above the tracheal bifurcation and the tracheoplasty site was rested. On postoperative day 15, the tube was changed to a tracheostomy one; 3 months postoperatively, no tracheostomy-related complications or rebleeding were observed. Therefore, VA-ECMO assisted tracheal repair is considered a useful treatment option for patients with tracheal disruption, where suturing a prosthesis to the tracheostomy stoma site is difficult.

Prophylactic innominate artery transection to prevent tracheoinnominate artery fistula: a retrospective review of single institution experiences.

PURPOSE: This study aimed to investigate the optimal indication and availability of prophylactic innominate artery transection (PIAT). METHODS: We retrospectively analyzed the medical records of the patients with neurological or neuromuscular disorders (NMDs) who underwent PIAT. Meanwhile, we originally defined the tracheal flatting ratio (TFR) and mediastinum-thoracic anteroposterior ratio (MTR) from preoperative chest computed tomography imaging and compared these parameters between non-PIAT and PIAT group. RESULTS: There were 13 patients who underwent PIAT. The median age was 22 years. PIAT was planned before in one, simultaneously in five, and after tracheostomy or laryngotracheal separation in seven patients. Image evaluations of the brain to assess circle of Willis were performed in all patients. Appropriate skin incisions with sternotomy to expose the innominate artery were made in four patients. All patients are still alive except one late death without any association with PIAT. No neurological complications occurred in any patients. As significant differences (p < 0.01) between two groups were observed for TFR and MTR, objective validity of the indication of PIAT was found. CONCLUSIONS: PIAT is safe and tolerable in case of innominate artery compression of the trachea with NMDs. TFR and MTR are useful objective indexes to judge the indication of PIAT.

Endovascular Treatment of Tracheoinnominate Artery Fistula: Case Report and Literature Review With Pooled Cohort Analysis.

To pool data from published cases of tracheoinnominate artery fistula (TIF) treated with surgical or endovascular techniques along with reporting a case of similar presentation. A total of 261 cases in 137 published case reports and case series were identified through a comprehensive systematic literature review. Data regarding patient characteristics, treatment, and follow-up were extracted. A local case of a 14-year-old boy with TIF due to longstanding tracheostomy treated with stent-graft placement was added to the data. Comparison of the complication rates between surgical vs endovascular interventions was done with the chi-square test. Factors associated with longer survival were assessed by the Cox regression analysis. Thirty-three (12.6%) of the reported cases were treated endovascularly, 137 (52.3%) were treated surgically, and 92 (35.1%) were reported with no definitive treatment. Mean age was 34 ± 22 years, and 61% were males. The mean time interval between tracheotomy placement and bleeding was 1 ± 2.5 years. A lower procedure-related complication (30% vs 50%, P = 0.045) and 30-day mortality (9% vs 23%, P = 0.008) rates had been reported with percutaneous approaches compared to surgery. No percutaneous procedure was reported prior to year 2000. In multivariate analysis stratified by publication year, a shorter tracheostomy-to-bleeding time (year) was significantly associated with higher hazards of death (hazard ratio: 1.22, P = 0.017). Type of intervention (percutaneous vs surgery) was not associated with postintervention survival (adjusted hazard ratio: 0.78, P = 0.558). Endovascular stent grafting can have a comparable postprocedural survival and lower complication rates vs open surgical repair in treatment of TIF.

Successful Rescue of a Ruptured Tracheoinnominate Fistula with Extracorporeal Membrane Oxygenation, Endovascular Stents, and Debranching Surgical Bypass.

We reported a case of ruptured tracheoinnominate fistula in a 14-year-old boy with history of repeated sternotomy. Tracheostomy was performed at age 2 years. Slide tracheoplasty was done at age 13 years. He presented to outpatient clinic with episodic hemosputum. Massive blood emanated from stoma during bronchoscopy evaluation. Venous-arterial extracorporeal membrane oxygenation was installed for resuscitation. A contrast-enhanced computed tomography (CT) and angiography confirmed the diagnosis. Immediate control of bleeding was achieved by an endovascular stent graft deployed at innominate artery. Massive hemorrhage recurred on day 7. An aortic arch stent was inserted and all arch vessels debranching via supraclavicular collar excision was performed. A covered stent was used to fenestrate the aortic stent and establish antegrade blood flow to all neck vessels via left common carotid artery. The patient remained stable at 10-month follow-up. Combination of extracorporeal membrane oxygenation, endovascular intervention, and surgical bypass could be effective in treating critical patients.

Tracheoinnominate Artery Fistula Formation in a Child with Long-Term Tracheostomy Dependence.

We report a fatal tracheoinnominate artery fistula (TIF) in a 13-year-old female patient with long-term tracheostomy tube dependence due to chronic respiratory failure. Thirteen years after placement of her tracheostomy tube, the patient experienced two separate episodes of sentinel bleeding prior to a fatal hemorrhagic event. Diagnostic evaluation after the sentinel events was mostly nonconclusive. This case highlights the risk of TIF in pediatric age group, even years after initial tracheostomy tube placement, and the need for a high index of suspicion for TIF when children present with unexplained tracheal bleeding.

Tracheostomy Emergencies.

Tracheostomy is a common procedure for long-term airway management. Although the overall complication rate is greater than 50%, the incidence of serious complications is low. These serious complications can, however, lead to significant morbidity and mortality and it is incumbent on the emergency provider to be prepared to deal with such tracheostomy-related emergencies. The greatest life threats to the tracheostomy patient are decannulation, obstruction, and hemorrhage. Other important but lower-acuity complications include tracheoesophageal fistula formation, tracheal stenosis, infection, and tracheocutaneous fistula formation.

Combined tracheoinnominate artery fistula and tracheoesophageal fistula: A very rare complication of indwelling tracheostomy tube.

Tracheoinnominate artery fistula (TIF) is a serious complication of tracheostomy. If untreated, it could be life-threatening. The emergency approach to the condition that includes prompt diagnosis, rapid control of bleeding with a clear airway, and operation with or without interruption of the innominate artery are the most important factors influencing patient outcome. Tracheoesophageal fistula (TEF) is another complication of tracheostomy. In association with compromised quality of life, this condition is really hard to be treated. We report a case of combined TIF and TEF in a 27-year-old man with quadriplegia who suffered a car accident but was successfully managed with interruption and ligature of the innominate artery repair of trachea.

Clinical outcomes of tracheoesophageal diversion and laryngotracheal separation for aspiration in patients with severe motor and intellectual disability.

CONCLUSIONS: Tracheoesophageal diversion (TED) and laryngotracheal separation (LTS) can prevent aspiration pneumonia and improve the morbidity of patients with severe motor and intellectual disability (SMID). By improving hospitalization rates and care needs, the quality-of-life can be improved for the patients and their parents. OBJECTIVES: This study evaluated the clinical outcomes of TED and LTS in patients with intractable aspiration and SMID. METHODS: This study retrospectively reviewed patients with SMID and intractable aspiration pneumonia who underwent TED or LTS at the institution between January 2008 and January 2015. It assessed the frequency of sputum suctioning, the number of pre-operative and post-operative hospitalizations, the operative time, and complications. RESULTS: Forty patients were identified during the study period. After surgery, there were significant reductions in the frequency of secretion suctioning (from 165.0 times/day to 33.0 times/day) and the number of hospitalizations because of aspiration pneumonia (from 5.4 times/year to 0.2 times/year). A tracheocutaneous fistula occurred in one (2.5%) patient, and two (5.4%) patients developed tracheoinnominate artery fistulas. In the latter group, the innominate arteries were successfully ligated and endovascular embolization was performed.

Recurrent Tracheoinnominate Artery Fistula due to Stent Graft Fracture.

Tracheoinnominate artery fistula (TIF) is one of the most dangerous complications after tracheostomy, and fetal even after surgical repair. Recently, endovascular stent has been introduced as an option for the treatment of TIF. Unfortunately, endovascular stent repair could not replace the surgery due to rare clinical reports about the long-term follow-up and complications of stent graft. More collection of clinical data are necessary for the evaluation of long-term results. We report a rare case of recurrent TIF due to stent graft fracture after endovascular stent graft insertion in the treatment of TIF, and reviewed the world literature.

Resection of the innominate artery to prevent an impending tracheoinnominate fistula and to permit tracheotomy in a patient with subglottic stenosis and high-riding innominate.

OBJECTIVE: This study aimed to determine the long-term viability of innominate artery resection and tracheotomy for a patient at high risk of developing a tracheoinnominate fistula (TIF) in the setting of subglottic stenosis and a high-riding innominate artery. METHODS: Chart review with 2-year follow-up. RESULTS: A 45-year-old diabetic man with obstructive sleep apnea and multiple admissions for coma and delirium tremens associated with alcohol abuse developed subglottic stenosis. He was found to have a palpable supraclavicular pulse during preoperative examination for a tracheotomy. Computed tomography examination revealed a high-riding innominate artery at the level of stenosis along with granulation tissue and disruption of the cartilaginous trachea, suggesting a high risk of impending TIF. The patient underwent a sternotomy-approach resection of the innominate artery with closure of the distal stump with a sternohyoid muscle flap. Intraoperatively, a plane of adhesions between the posterior innominate artery and trachea was dissected. The anterior tracheal wall appeared calcified but without evidence of erosion of either the trachea or the artery. Six weeks later, a tracheotomy was performed. Follow-up at 27 months did not identify complications from the innominate artery resection. CONCLUSION: Resection of the innominate artery is an option for some patients either to address the warning signs of TIF or to permit a tracheotomy to be performed in the presence of a high innominate artery.

Innominate artery compression of the trachea in patients with neurological or neuromuscular disorders.

OBJECTIVES: The purpose of this study was to review and assess our surgical management of innominate artery compression of the trachea (IACT) in patients with neurological or neuromuscular disorders (NMDs). METHODS: Thirty patients with NMD who underwent surgical treatment for IACT at Kobe Children's Hospital and Kobe University Hospital from 2002 to 2012, were enrolled in this retrospective study. The clinical outcomes of preventive elective surgery for IACT (Group A, n=20) were assessed and compared with those of emergent surgery (Group B, n=10). RESULTS: A total of 27 patients underwent innominate artery transection (17 in Group A and 10 in Group B), and 3 patients in Group A underwent innominate artery reimplantation using prosthetic graft interposition. No operative or early death occurred. There were no cases of postoperative mediastinitis or neurological complications. The operative benefits in Group A included a smaller skin incision, more limited sternotomy, less blood loss, shorter operative time and shorter hospital stay, compared with Group B. No blood transfusion was required in Group A. The number of patients in whom cerebral circulation was assessed before surgery in Group A was significantly higher than those in Group B. CONCLUSIONS: Preventive elective surgery for IACT provides many advantages, including minimally invasive procedures and successful postoperative outcomes without neurological complications in patients with NMD. Because this surgical management can prevent the tragic occurrence of a tracheo-innominate artery fistula or an exacerbation of tracheomalacia, it would be an optimal surgical treatment for IACT to improve the quality of life in patients with NMD.

Successful treatment of tracheoinnominate artery fistula following tracheostomy in a patient with cerebrovascular disease.

Tracheoinnominate artery fistula is a critical complication of tracheostomy. The most important factors influencing patient outcome are prompt diagnosis, immediate control of bleeding with a patent airway, and emergency operation with or without interruption of the innominate artery. Here, we report a case of tracheoinnominate artery fistula in a 40-year-old woman with cerebrovascular accident who was successfully managed with an aorta-axillary artery bypass.

Successful Treatment of Tracheoinnominate Artery Fistula Following Tracheostomy in a Patient with Cerebrovascular Disease

Tracheoinnominate artery fistula is a critical complication of tracheostomy. The most important factors influencing patient outcome are prompt diagnosis, immediate control of bleeding with a patent airway, and emergency operation with or without interruption of the innominate artery. Here, we report a case of tracheoinnominate artery fistula in a 40-year-old woman with cerebrovascular accident who was successfully managed with an aorta-axillary artery bypass.

Is computerized tomography angiographic surveillance valuable for prevention of tracheoinnominate artery fistula, a life-threatening complication after tracheostomy?

OBJECTIVE: The aim of this study was to evaluate the utility of volume-rendered helical computerized tomography (CT) angiography focusing tracheostomy tube and innominate artery for prevention of tracheoinnominate artery fistula. METHODS: The authors retrospectively analyzed 22 patients with tracheostomy who had checked CT angiography. To evaluate the relationship between tracheostomy tube and innominate artery, we divided into three categories. First, proximal tube position based on cervical vertebra, named "tracheostomy tube departure level (TTDL)". Second, distal tube position and course of innominate artery, named "tracheostomy tube-innominate artery configuration (TTIC)". Third, the gap between the tube and innominate artery, named "tracheostomy tube to innominate artery gap (TTIG)". The TTDL/TTIC and TTIG are based on 3-dimensional (3D) reconstruction around tracheostomy and enhanced axial slices of upper chest, respectively. RESULTS: First, mean TTDL was 6.8±0.6. Five cases (23%) were lower than C7 vertebra. Second, TTIC were remote to innominate artery (2 cases; 9.1%), matched with it (14 cases; 63.6%) or crossed it (6 cases; 27.3%). Only 9% of cases were definitely free from innominate artery injury. Third, average TTIG was 4.3±4.6 mm. Surprisingly, in 6 cases (27.3%), innominate artery, trachea wall and tracheostomy tube were tightly attached all together, thus have much higher probability of erosion. CONCLUSION: If low TTDL, match or crossing type TTIC with reverse-L shaped innominate artery, small trachea and thin TTIG are accompanied all together, we may seriously consider early plugging and tube removal.

Is Computerized Tomography Angiographic Surveillance Valuable for Prevention of Tracheoinnominate Artery Fistula, a Life-Threatening Complication after Tracheostomy?

OBJECTIVE: The aim of this study was to evaluate the utility of volume-rendered helical computerized tomography (CT) angiography focusing tracheostomy tube and innominate artery for prevention of tracheoinnominate artery fistula. METHODS: The authors retrospectively analyzed 22 patients with tracheostomy who had checked CT angiography. To evaluate the relationship between tracheostomy tube and innominate artery, we divided into three categories. First, proximal tube position based on cervical vertebra, named "tracheostomy tube departure level (TTDL)". Second, distal tube position and course of innominate artery, named "tracheostomy tube-innominate artery configuration (TTIC)". Third, the gap between the tube and innominate artery, named "tracheostomy tube to innominate artery gap (TTIG)". The TTDL/TTIC and TTIG are based on 3-dimensional (3D) reconstruction around tracheostomy and enhanced axial slices of upper chest, respectively. RESULTS: First, mean TTDL was 6.8+/-0.6. Five cases (23%) were lower than C7 vertebra. Second, TTIC were remote to innominate artery (2 cases; 9.1%), matched with it (14 cases; 63.6%) or crossed it (6 cases; 27.3%). Only 9% of cases were definitely free from innominate artery injury. Third, average TTIG was 4.3+/-4.6 mm. Surprisingly, in 6 cases (27.3%), innominate artery, trachea wall and tracheostomy tube were tightly attached all together, thus have much higher probability of erosion. CONCLUSION: If low TTDL, match or crossing type TTIC with reverse-L shaped innominate artery, small trachea and thin TTIG are accompanied all together, we may seriously consider early plugging and tube removal.