Spontaneous aberrant right subclavian arterio-oesophageal fistula in a previously healthy child

Joynt, Michael; Grifka, Ronald G.

doi:10.1017/s1047951114002388

Cited by 4 publications

(5 citation statements)

References 9 publications

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“…13,20 Six cases were in the pediatric population, with patient ages ranging from 5 months to 11 years. 22,28,30,34,37 Although diagnosis is typically made with CT imaging, endoscopic visualization may be the initial diagnostic test to exclude other causes, particularly in patients who are hemodynamically stable at the time of presentation. The most common etiology for development of ARSA-esophageal fistula appeared to be secondary to compression, friction, or pressure necrosis from recent instrumentation with an endotracheal tube, nasogastric tube, or tracheostomy tube, seen in 14 of the 33 summarized cases.…”

Section: Discussionmentioning

confidence: 99%

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Upper Gastrointestinal Bleeding From Aberrant Right Subclavian Artery-Esophageal Fistula

Peress

Tuqan

Thomas

2021

TOJ

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Background:The most common aortic arch abnormality is an aberrant right subclavian artery (ARSA). ARSA-esophageal fistula is a rare sequela that can present with a life-threatening upper gastrointestinal (GI) bleed. Case Report: We report the case of an 88-year-old male who presented with signs of upper GI bleeding. Esophagogastroduodenoscopy demonstrated extrinsic compression of the upper third of the esophagus with ulceration. Imaging studies revealed ARSA posterior to the esophagus with pseudoaneurysm formation. These findings confirmed an upper GI bleed secondary to ARSA-esophageal fistula. The patient underwent prompt embolization of the ARSA pseudoaneurysm, followed a few days later by coil embolization of the ARSA pseudoaneurysm. Despite these interventions, the patient continued to have bleeding with anemia. He and his family opted to avoid any further interventions and instead pursued comfort care. The patient was discharged to hospice and died 3 months later. Conclusion: ARSA-esophageal fistula is a rare but potentially lethal cause of upper GI bleeding. Initial signs and symptoms can be subtle, but the presence of a GI bleed requires immediate stabilization. Surgical interventions have been shown to have longerlasting success, but endovascular repair may be an option for patients who are deemed unfit for surgery but still require prompt stabilization. Regardless of the intervention, mortality rates for ARSA-esophageal fistula are high.

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Section: Discussionmentioning

confidence: 99%

“…Of the 33 summarized cases, thirteen cases survived all episodes of bleeding (39%). 21,22,24,27,28,30,31,33,34,[36][37][38]43 The severity of bleeding, in the setting of several comorbid conditions, likely contributed to the high mortality rate.…”

Section: Discussionmentioning

confidence: 99%

Upper Gastrointestinal Bleeding From Aberrant Right Subclavian Artery-Esophageal Fistula

Peress

Tuqan

Thomas

2021

TOJ

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“…Eleven cases of nonaortic AEF have been reported in recent 20 years (1997–2017). They were mainly male and middle-aged (30s to 70s-year-old), except for a 17-month-old female [1] . The responsible artery for nonaortic AEF included aberrant right subclavian artery (ARSA), bronchial artery, broncho-intercostal arterial trunk, intercostal artery, and left subclavian artery.…”

Section: Discussionmentioning

confidence: 99%

“…Eventually, AEF is formed. The second category is vascular surgery and thoracic arterial malformations which includes ARSA [1] . The ARSA is a rare anomaly and happened in about 0.5%–2.0% of the population [8] .…”

Section: Discussionmentioning

confidence: 99%

Massive upper gastrointestinal bleeding caused by an intercostal arterio-esophageal fistula: A rare case report

Yang

Lin

Huang

et al. 2020

Radiology Case Reports

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Arterio-esophageal fistula (AEF) is a rare life-threatening cause of upper gastrointestinal bleeding. Realizing the risk factors and clinical presentations of AEF will enable us to provide timely diagnosis, efficient treatment, and better outcome. We present a 43-year-old Taiwanese man who had histories of destructive lung, innominate artery-trachea fistula, and received bilobectomy. He was admitted to our hospital with fresh blood drainage from nasogastric tube. He was diagnosed as right fifth intercostal artery esophageal fistula by computed tomography angiography and transcatheter arterial embolization was performed. The bleeding stopped and the patient was discharged uneventfully. The risk factors of AEF include esophageal foreign body, vascular surgery, thoracic arterial malformations, and concurrent chemoradiotherapy in T4-esophageal cancer. Computed tomography angiography may be the most sensitive diagnostic test. Transcatheter arterial embolization or endovascular stent grafting may be considered as the first-line treatment of AEF in the future.

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“…Although cases of so-called spontaneous fistula have been reported, pressure from these devices or even an ingested foreign body may result in necrosis of adjacent tissue leading to fistula formation. 16 Due to association of EA and ARSA, perhaps it is not surprising that ARSA-esophageal fistula is seen in the pediatric and young adult population. There are eight reported cases in patients younger than 20 years, with four having either EA or TEF.…”

Section: Discussionmentioning

confidence: 99%

Aberrant Right Subclavian Artery-Esophageal Fistula in 20-Year-Old with VATER Association

Kudose

Pineda

Saito

et al. 2016

J Pediatr Intensive Care

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Aberrant right subclavian artery (ARSA), the most common aortic arch abnormality, occurs in approximately 0.5 to 1.8% of the general population, with prevalence of up to 25% in those with esophageal atresia. Although ARSA is often asymptomatic, a fistulous tract into esophagus may develop with prolonged nasogastric tube placement or endotracheal intubation and lead to potentially fatal hematemesis. We present a first case of ARSA-esophageal fistula in a 20-year-old woman with VATER association in the absence of an esophageal anomaly and review 28 cases of ARSA-esophageal fistula reported in the literature to date. Requiring nasogastric and endotracheal tube placement for approximately 4 months, the patient had a prolonged hospital course and died after sudden hematemesis. An autopsy demonstrated an ARSA-esophageal fistula and no other source of upper gastrointestinal bleeding. In patients with esophageal atresia requiring prolonged placement of an endotracheal or nasogastric tube, a screening imaging study and corrective surgery may be indicated. Although the mortality rate is still high, timely recognition and repair of ARSA-esophageal fistula appear to be improving. Given the potentially prolonged latency for its development with occasional presence of heralding symptoms, increased awareness may facilitate surgical intervention to prevent a catastrophic exsanguination.

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Spontaneous aberrant right subclavian arterio-oesophageal fistula in a previously healthy child

Cited by 4 publications

References 9 publications

Upper Gastrointestinal Bleeding From Aberrant Right Subclavian Artery-Esophageal Fistula

Upper Gastrointestinal Bleeding From Aberrant Right Subclavian Artery-Esophageal Fistula

Massive upper gastrointestinal bleeding caused by an intercostal arterio-esophageal fistula: A rare case report

Aberrant Right Subclavian Artery-Esophageal Fistula in 20-Year-Old with VATER Association

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