Fatal aortoesophageal fistula in two cases of tight vascular ring

2002 ◽  
Vol 12 (2) ◽  
pp. 172-176 ◽  
Author(s):  
Annalisa Angelini ◽  
Konstantinos Dimopoulos ◽  
Carla Frescura ◽  
Piergiorgio Gamba ◽  
Roberto Lo Piccolo ◽  
...  

Vascular rings are rare vascular congenital anomalies causing oesophageal and tracheal compression. An aortoesophageal fistula is a devastating, in part iatrogenic, complication of vascular rings. It is seen with increasing frequency, and can be misleading, since differential diagnosis with other causes of haematemesis and melaena is often difficult, especially in infants. We report two infants with aortoesophageal fistulas secondary to double aortic arches forming a vascular ring. In both, the diagnosis was missed, and massive haemorrhage led to death. In both cases, the fissuration on the oesophageal and aortic sides of the fistula had sharp edges, highly suggestive of an iatrogenic laceration caused by manipulation of nasogastric tubes. The key for the diagnosis of vascular rings is, therefore, clinical suspicion and awareness of this condition. Prompt identification in infants with stridor, wheezing, or respiratory distress can prevent prolonged intubation, thus avoiding the formation of an aortoesophageal fistula and hopefully preventing a fatal outcome.

2011 ◽  
Vol 125 (11) ◽  
pp. 1158-1163 ◽  
Author(s):  
E Phelan ◽  
S Ryan ◽  
H Rowley

AbstractIntroduction:A vascular ring refers to encirclement of the trachea and oesophagus by an abnormal combination of derivatives of the aortic arch system. These malformations can cause variable degrees of compression of the oesophagus, trachea or both. Symptoms can range from no effect to severe stridor, dyspnoea and/or dysphagia.Method and results:This study presents a case series of six patients treated over a six-year period (2003–2009), illustrating the features of four different types of vascular ring; these types are discussed in detail. The clinical presentation, radiology, and microlaryngoscopy and bronchoscopy findings are also discussed.Conclusion:The management of children with vascular rings requires a high index of clinical suspicion to ensure prompt diagnosis. As many of these children present with airway symptoms, the paediatric otolaryngologist plays a key role in identifying and assessing their anatomical anomalies.


2020 ◽  
Vol 41 (Supplement_2) ◽  
Author(s):  
G Amir ◽  
N Soffair ◽  
G Frenkel ◽  
E Bruckheimer ◽  
E Nachum ◽  
...  

Abstract Background Vascular anomalies of the Aortic arch can cause respiratory symptoms in children due to tracheal compression. Treatment consists of division of the vascular rings, nevertheless data regarding mid- term results is scarce. The purpose of this study was to evaluate clinical results of vascular ring surgery. Methods Between2007–2014, 85 children underwent vascular ring surgery. 51 had Double Aortic Arch (DAA, 60%), 31 Right Arch with Aberrant Subclavian Artery (RAA & ALSA, 36.5%) and 3 had RAA, mirror image branching & left ductus arteriosus (3.5%). Mean age and weight at operation were 12.4±13months and 8.6±4.1 kg respectively. Mid-term follow-up included clinical follow up by a pulmonologist (38 patients, 44.7%) and a telephone questionnaire (71 patients, 83%) performed 57±25.7 months after surgery. Results In most patients, symptomatic relief occurred in less than 6 months (table 1). Mid –term follow up revealed that although most parents described a significant improvement in their child's respiratory symptoms (95%), a significant number of patients described some residual respiratory symptoms (table 2). We did not find any significant association between age at surgery (under 6 month), or vascular ring anatomy (DAA vs. RAA&ASA) and the presence of residual symptoms at follow up Conclusions Surgical division of vascular rings results in a significant clinical improvement within one year, nevertheless many patients remain symptomatic to some degree. We found no association between the age at surgery or anatomic variant to the presence of symptoms in mid- term follow up. Further evaluation whether a more aggressive surgical approach is warranted in order to decrease the incidence long-term symptoms. Funding Acknowledgement Type of funding source: None


2018 ◽  
Vol 36 (2) ◽  
pp. 343.e1-343.e3 ◽  
Author(s):  
Yang Yang ◽  
Die Hu ◽  
Daoquan Peng

2002 ◽  
Vol 81 (8) ◽  
pp. 554-555 ◽  
Author(s):  
Liam J. Skinner ◽  
Stephanie Ryan ◽  
John D. Russell

The diagnosis of a vascular ring can be made on the basis of characteristic findings on barium esophagography. We report a case of a double aortic arch in a 9-month-old girl that was diagnosed in this manner, and we briefly review the anatomic characteristics of vascular rings.


2005 ◽  
Vol 71 (4) ◽  
pp. 289-291 ◽  
Author(s):  
Curt S. Koontz ◽  
Amina Bhatia ◽  
Joe Forbess ◽  
Mark L. Wulkan

Vascular rings are usually repaired via left thoracotomy. We report our series of pediatric patients with vascular rings that were repaired thoracoscopically. From February 2002 to September 2004, 13 patients underwent video-assisted thoracoscopic surgical techniques (VATS) division of their vascular ring. Chest magnetic resonance arterography (MRA) and/or computed tomographic arteriography (CTA) were used to evaluate the vascular ring in most patients. Patients were chosen for VATS repair based on surgeon's choice and type of vascular ring. Data are expressed as mean ± SD. The Children's Healthcare of Atlanta Institutional Review Board approved this retrospective chart review. Age and weight was 1.5 ± 1.8 years (range: 4 months–17 years) and 16.0 ± 12.5 kg (range: 6.0–22.1 kg), respectively (n = 13). Associated diseases included congenital heart disease (n = 2). Symptoms included respiratory complaints (n = 6), dysphagia (n = 2), dysphagia and shortness of breath (n = 1), pneumonia (n = 2), tracheal deviation (n = 1), and one patient was asymptomatic. Vascular ring types included double aortic arch (n = 4) and right aortic arch with an aberrant left subclavian artery and a left ligamentum arteriosum (n = 9). Operating time was 70 ± 20 minutes (range: 46–122 minutes). One patient had to be opened because of a large arch. Length of stay was 1.9 ± 0.9 days (range: 1–3 days). There were no complications, and all patients improved clinically at follow-up. Thoracoscopic repair of certain types of vascular rings seems to be safe and effective in children. More patients, however, need to be studied.


2020 ◽  
Vol 21 (Supplement_1) ◽  
Author(s):  
I Campos ◽  
C Vieira ◽  
N Salome ◽  
V H Pereira ◽  
A Costeira Pereira ◽  
...  

Abstract Introduction Complete vascular rings represent about 0.5-2% of all congenital cardiovascular malformations, with the double aortic arch (DAA) being the most common of the complete vascular rings, causing tracheoesophageal compression. The right (posterior) arch is usually dominant (70%), although the two arches can have the same size (5%). The left (anterior) arch is dominant in only approximately 25% of cases. In most cases, this anomaly is diagnosed during childhood due to symptoms caused by oesophageal or tracheal compression. For this reason, case reports of adults are rare. This report describes a case of a 61-year-old woman with DAA with dominant left arch, diagnosed accidentallyby thoracic CT angiography. Case Report Description A 61 years old woman with a previous story of hypertension and type 1 diabetes presented to the emergency service with dyspnoea and thoracic pain. She also referred a history of intermittent dysphagia and cough with at least 12 years of progression. All the parameters of the physical examination were within normal limits. The electrocardiogram showed a normal sinus rhythm with no evidence of acute ischemia and her blood analyses did not show any abnormalitie. She also performed a thoracic CT angiography, which excluded signs of pulmonary embolism, but revealed a vascular ring suggesting a double aortic arch with permeability in both right and left arches as well as their collaterals. The Cardiac MRI was performed with the purpose of excluding ischemia, confirming the double aortic arch with left dominance. The right arch, posterior to the oesophagus and trachea, and the left arch, in an anterior position, showed an anatomic compression of the oesophagus as well as the proximal trachea, capable of eliciting the symptoms mentioned. Other congenital anomalies were excluded. The echocardiography did not demonstrate any additional cardiac malformation. Endoscopy shows a pulsatile extrinsic compression of the esophagus (aortic ring). The patient is currently being studied and closely monitored in the Cardiology consultation. Discussion The most common type of complete vascular ring is the double aortic arch, which accounts for 70% of the complete rings. In most cases, there are two permeable arches, usually with right dominance (70% of the cases). Rarely, both arches are symmetrical. Symptoms usually appear in the fifth month of life. In most cases, only supportive treatment is required. Conclusion This case illustrates the atypical features of this congenital malformation, namely the diagnosis during adulthood as well as the left dominance. Abstract P725 Figure. A double aortic arch


Author(s):  
Tzu-Ching Shih ◽  
Tzyy-Leng Horng ◽  
Fong-Lin Chen

Vascular rings, congenital intracardic anomalies of the aortic arch and the vessels emerging from the heart, completely encircle the trachea and esophagus [1]. The vascular ring results in narrowing and obstruction of the trachea and the esophagus. Due to the existence of a complete or partial vascular ring compressing either the trachea or esophagus, symptoms of a vascular ring in children include cough, stridor, chronic cough, dysphagia, persistent wheeze, and noisy breathing [2]. Some studies reported that the vascular ring surgery provides an excellent chance to improve the patient respiration conditions, especially for relief of symptoms [1–3]. Al-Bassam et al. reported that the thoracoscopic division of vascular rings in infants and children is a safe and effective surgery rather than an open thoracotomy[4]. Even after the treatment of a surgical division of the vascular ring, however, the fixed obstruction is relieved but the patient continues to have dynamic collapse because the compressed trachea segment is always malacic. Airway resistance to flow in the airway, thus, is a key factor for not only clinical diagnosis severity assessment but also therapeutic decision in tracheal stenosis. Furthermore, Malvè et al. (2011) utilized the finite element-based commercial software code (ADINA R&D Inc.) to model the fluid structure interaction of a human trachea under different ventilation conditions [5]. They also found that the positive pressure in the trachea does not result in the airway collapse during the time period of mechanical breathing. Therefore, the purpose of this study is to use the computational fluid dynamics (CFD) technique to calculate the local pressure drops in the tracheal segment for different inspiratory and expiratory flow rates due to preoperative and preoperative vascular ring surgery.


2021 ◽  
Vol 25 (1) ◽  
pp. 39-44
Author(s):  
Venus Chegini ◽  
◽  
Victoria Chegini ◽  
Mahdis Esfahani ◽  
Hossein Moeini ◽  
...  

Background: Subglottic stenosis is among the most common airway problems in children, i.e., acquired or congenital. More than 90% of acquired cases are secondary to endotracheal intubation and occur iatrogenically. Subglottic stenosis is an unexpected problem that requires timely diagnosis and intervention. Most cases of subglottic stenosis in children are mild to moderate. Case Presentation: The presented patient was a 10-year-old child who, after long intubation due to head trauma following early discharge from the surgical center, was referred to the Children’s Hospital on the same day with a complaint of high fever and shortness of breath. With the development of respiratory distress and cyanosis, the patient was transferred to the intensive care unit. Due to the impossibility of passing the tracheal tube with the appropriate age for the patient, the initial diagnosis of subglottic stenosis was established and a tracheostomy was performed. Diagnostic bronchoscopy confirmed severe subglottic stenosis, and the child was referred to a specialized Ear, Nose, Throat (ENT) center for the repair of the tracheal stenosis. Conclusion: One of the most common causes of stridor in children is subglottic stenosis following prolonged intubation. Recognizing the predisposing factors, prevention, strong clinical suspicion, timely diagnosis, and treatment can prevent further adverse complications or consequences in children.


2019 ◽  
Vol 11 (1) ◽  
pp. 120-122 ◽  
Author(s):  
Maris Bartkevics ◽  
Zoran Stankovic ◽  
Susanne Schibli ◽  
Simon Fluri ◽  
Steffen Berger ◽  
...  

We report a case of an infant surviving aortoesophageal fistula secondary to lithium cell battery ingestion. In the setting of a delayed vascular complication, computed tomography and magnetic resonance imaging are essential to establishing the correct diagnosis and surgical management. Management of children after battery ingestion must be guided by a high index of clinical suspicion.


2020 ◽  
pp. 004947552095990
Author(s):  
Şule Gökçe ◽  
Nüsabe Abdullayeva ◽  
Feyza Koç ◽  
Hüseyin Hüdaver Alper

Vascular rings are rare anomalies that occur as a result of abnormal development of the aortic arch complex, often manifested by signs of tracheo-oesophageal compression during the infant period. A double aortic arch anomaly is the most common type of vascular ring pathology. Here we report a case presenting with wheezing and stridor in a 10-month-old child.


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