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      Aortopexy for the treatment of tracheomalacia in children: review of the literature

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          Abstract

          Abstract

          Severe tracheomalacia presents a significant challenge for Paediatricians, Intensivists, Respiratory Physicians, Otolaryngologists and Paediatric Surgeons. The treatment of tracheomacia remains controversial, but aortopexy is considered by most to be one of the best options. We conducted a review of the English literature relating to aortopexy.

          Among 125 papers, 40 have been included in this review. Among 758 patients (62% males) affected with tracheomalacia, 581 underwent aortopexy. Associated co-morbidities were reported in 659 patients. The most frequent association was with oesophageal atresia (44%), vascular ring or large vessel anomalies (18%) and innominate artery compression (16%); in 9% tracheomalacia was idiopathic. The symptoms reported were various, but the most important indication for aortopexy was an acute life-threatening event (ALTE), observed in 43% of patients. The main preoperative investigation was bronchoscopy. Surgical approach was through a left anterior thoracotomy in 72% of patients, while median approach was chosen in 14% and in 1.3% a thoracoscopic aortopexy was performed. At follow-up (median 47 months) more than 80% of the patients improved significantly, but 8% showed no improvement, 4% had a worsening of their symptoms and 6% died. Complications were observed in 15% of patients, in 1% a redo aortopexy was deemed necessary.

          In our review, we found a lack of general consensus about symptom description and evaluation, indications for surgery, though ALTE and bronchoscopy were considered by all an absolute indication to aortopexy and the gold standard for the diagnosis of tracheomalacia, respectively. Differences were reported also in surgical approaches and technical details, so that the same term “aortopexy” was used to describe different types of procedures. Whatever approach or technique was used, the efficacy of aortopexy was reported as high in the majority of cases (more than 80%). A subgroup of patients particularly delicate is represented by those with associated gastro-esophageal reflux, in whom a fundoplication should be performed. Other treatments of tracheomalacia, particularly tracheal stenting, were associated with a higher rate of failure, severe morbidity and mortality.

          Non english abstract

          La tracheomalacia severa rappresenta una sfida per Pediatri, Intensivisti, Pneumologi, Otorinolaringoiatri, Chirurghi Pediatri. Il trattamento della tracheomalacia è tuttora controverso. L’aortopessi è considerata da molti la migliore opzione terapeutica. Abbiamo condotto una revisione della letteratura di lingua inglese su tale argomento.

          Di 125 lavori, 40 sono stati inclusi nella revisione. Tra 758 pazienti (62% maschi) affetti da tracheomalacia, 581 sono stati sottoposti ad aortopessi tra il 1968 e il 2008. In 659 pazienti alcune comorbidità erano presenti. L’associazione più frequente era con l’atresia esofagea (44%), l’anello vascolare o un’anomalia dei grossi vasi (18%), la compressione da parte dell’arteria innominata (16%); nel 9% la tracheomalacia era idiopatica. I sintomi riportati sono stati variabili, ma l’indicazione più importante all’aortopessi sono stati eventi di ALTE, osservati nel 43% dei pazienti. Lo studio diagnostico preoperatorio principale è stato la broncoscopia. L’approccio chirurgico è avvenuto attraverso una toracotomia anteriore sinistra nel 72% dei pazienti, mentre un approccio mediano è stato scelto nel 14% e nell’1.3% dei casi è stato eseguito un approccio toracoscopico. Al follow-up (mediana di 47 mesi) la maggioranza dei pazienti sono migliorati significativamente, ma l’8% di essi non è migliorato, il 4% è peggiorato e il 6% è morto. Complicazioni sono state riportate nel 15% dei pazienti, nell’1% un nuovo intervento di aortopessi è stato necessario.

          In questa revisione abbiamo trovato che non c’è un consenso generale sulla valutazione e sulla descrizione dei sintomi, sulle indicazioni chirurgiche ed esami preoperatori, anche se le ALTE e la broncoscopia venivano considerate rispettivamente un’indicazione assoluta all’aortopessi e il “gold standard” diagnostico per la tracheomalacia. Venivano riportate differenze negli approcci chirurgici e nei dettagli tecnici, e lo stesso termine di aortopessi veniva usato per indicare diverse procedure chirurgiche. In ogni caso, indipendentemente dall’approccio o tecnica utilizzati, l’efficacia dell’aortopessi veniva riportata come elevata nella maggioranza dei casi (più dell’80%). Un sottogruppo di pazienti particolarmente delicato è rappresentato da quelli con reflusso gastroesofageo associato, nei quali sarebbe indicata una fundoplicatio. Altri trattamenti della tracheomalacia, quali stent tracheale, sembrano gravati da una maggiore percentuale di insuccessi, morbidità severa e mortalità.

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          Most cited references11

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          Computed tomography versus bronchography in the diagnosis and management of tracheobronchomalacia in ventilator dependent infants.

          To assess the relative accuracy of dynamic spiral computed tomography (CT) compared with tracheobronchography, in a population of ventilator dependent infants with suspected tracheobroncho-malacia (TBM). Paediatric intensive care unit in a tertiary teaching hospital. Infants referred for investigation and management of ventilator dependence and suspected of having TBM were recruited into the study. Tracheobronchography and CT were performed during the same admission by different investigators who were blinded to the results of the other investigation. The study was approved by the hospital research ethics committee, and signed parental consent was obtained. Sixteen infants were recruited into the study. Fifteen had been born prematurely, and five had cardiovascular malformations. In 10 patients there was good or partial correlation between the two investigations, but in six patients there was poor or no correlation. Bronchography consistently showed more dynamic abnormalities, although CT picked up an unsuspected double aortic arch. Radiation doses were 0.27-2.47 mSv with bronchography and 0.86-10.67 mSv with CT. Bronchography was a better investigation for diagnosing TBM and in determining opening pressures. Spiral CT is unreliable in the assessment of TBM in ventilator dependent infants. In addition, radiation doses were considerably higher with CT.
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            Retroperitoneal cystic lymphangioma: a rare presentation in childhood, treated laparoscopically.

            Retroperitoneal cystic lymphangioma is a rare benign lesion of childhood. A 15-year-old girl underwent laparoscopic excision of such a cyst. On histopathological examination, the resection was complete. She had an uneventful recovery and remains asymptomatic. We report this in view of the rarity of this condition and also the unusual presentation of this case. Complete surgical excision via laparoscopy is a feasible option.
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              Aortic suspension (aortopexy) for severe tracheomalacia in infants and children.

              Tracheomalacia with anterior great vessel compression is a common disorder in infants and children, which can lead to life-threatening airway occlusion. In this study, a large number of patients underwent anterior aortopexy to provide a more normal distal airway. Thirty-two infants and children with tracheomalacia associated with esophageal atresia-tracheoesophageal fistula (18), vascular ring (8), abnormal innominate artery takeoff (4), and primary (2) were evaluated with bronchoscopy, magnetic resonance imaging, and pulmonary functions. Aortopexy was accomplished through left thoracotomy and suture fixation of the aorta and innominate artery to the posterior sternum. Intraoperative bronchoscopy showed marked improvement in airway caliber and rigidity, and no patients had further obstructive episodes. Forced expiratory volume improved from 52% +/- 4% of predicted to 82% +/- 3%. Aortopexy is a simple procedure for the treatment of distal tracheomalacia that is immediately effective and provides permanent relief of obstructive episodes.
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                Author and article information

                Journal
                Ital J Pediatr
                Ital J Pediatr
                Italian Journal of Pediatrics
                BioMed Central
                1824-7288
                2012
                30 October 2012
                : 38
                : 62
                Affiliations
                [1 ]Paediatric Surgery, G. Gaslini Institute, Genova, 16145, Italy
                [2 ]The National Service for Severe tracheal Disease in Children, The Great Ormond Street Hospital for Children NHS Trust, London, WC1N 3JH, United Kingdom
                Article
                1824-7288-38-62
                10.1186/1824-7288-38-62
                3502176
                23110796
                068f7c90-d36a-44e4-ae72-547182577b27
                Copyright ©2012 Torre et al.; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 16 May 2012
                : 4 October 2012
                Categories
                Review

                Pediatrics
                bronchoscopy,sternotomy,tracheal stent,gastro-oesophageal reflux,tracheomalacia,alte,thoracotomy,aortopexy,oesophageal atresia,thoracoscopy

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