Arterioesophageal fistula secondary to rupture of an aberrant right subclavian artery aneurysm: a rare differential diagnosis in upper gastrointestinal bleeding

Lehmann, Beat; Clemetson, I.; Fantin, Alberto; Henning, P. Troy; Kipfer, Beat; Mühlethaler, R.; Vetsch, G.; Dinkel, Hans-Peter

doi:10.1055/s-2006-925172

Cited by 7 publications

(5 citation statements)

References 3 publications

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“…Only 10 other cases attributed to this etiology have been reported. 11,12,16,18,19,23,25,33,35,42 Only 1 patient with upper GI bleed secondary to ARSA aneurysm survived all bleeding episodes, and the patient required balloon tamponade with surgery to replace the aorta. 33 Our patient survived the initial bleeding event, but died 3 months later, as he declined definitive surgical intervention to stop the recurrent bleeding.…”

Section: Discussionmentioning

confidence: 99%

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%

Upper Gastrointestinal Bleeding From Aberrant Right Subclavian Artery-Esophageal Fistula

Peress

Tuqan

Thomas

2021

TOJ

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“…Using the search terms “aneurysm” and “aberrant right subclavian artery,” and “rupture,” we found 24 entries. Of these, only two referred to ruptured aneurysms, 8 , 9 and only one of these underwent repair. Twelve articles described hybrid or endovascular treatments for aberrant right subclavian artery aneurysms, 1 , 5 , 6 , 10 , 11 , 12 , 13 , 14 , 15 and the other 10 were unrelated to our topic.…”

Section: Discussionmentioning

confidence: 99%

Hybrid management of a ruptured right subclavian artery aneurysm dissection

Drullinsky

Gill

Bayne

et al. 2017

Journal of Vascular Surgery Cases, Innovations and Techniques

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Aberrant right subclavian artery is the most common congenital malformation of the aortic arch (0.4%-2.0%). Aneurysms of aberrant subclavian arteries are extremely rare. This results in little experience with their treatment. We describe a case of a patient who presented to the emergency department with a dissection of an aberrant right subclavian artery that later progressed to rupture. Besides hemodynamic instability, this caused an acute superior vena cava syndrome, making airway control difficult. In the operating room, we obtained proximal control through thoracic endovascular aortic repair; median sternotomy was performed for distal control and evacuation of massive hemomediastinum.

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“…No gender predilection was noted in our review, but others have reported a female predilection. 4 Instrumentation within the adjacent trachea or esophagus is a suspected risk factor; 16 cases reported various forms of instrumentations such as endotracheal intubation, tracheostomy, or placement of a nasogastric tube. 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.…”

Section: Discussionmentioning

confidence: 99%

“…Importantly, mortality after massive bleeding in unstable patients remains much higher (74%). [4][5][6][7][8][9][10][11][12]14,17,18,[20][21][22][24][25][26][27] Pathologic findings when described show attenuation and necrosis of the arterial and esophageal wall with a variable inflammatory response. In two cases, the presence of neutrophils was accompanied by "polymicrobial bacterial overgrowth" and gram-positive cocci and Candida were identified, suggesting a potential role for infectious organisms.…”

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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Arterioesophageal fistula secondary to rupture of an aberrant right subclavian artery aneurysm: a rare differential diagnosis in upper gastrointestinal bleeding

Cited by 7 publications

References 3 publications

Upper Gastrointestinal Bleeding From Aberrant Right Subclavian Artery-Esophageal Fistula

Upper Gastrointestinal Bleeding From Aberrant Right Subclavian Artery-Esophageal Fistula

Hybrid management of a ruptured right subclavian artery aneurysm dissection

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

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