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Arterioesophageal Fistula: A Rare Complication of Retroesophageal Subclavian Arteries
Arterioesophageal Fistula: A Rare Complication of Retroesophageal Subclavian Arteries Patrick Feugier, MD,1 Laurent Lemoine, MD,1 Laurent Gruner, MD,2 Marc Bertin-Maghit, MD,3 Bernard Rousselet, MD,3 and Jean-Michel Chevalier, MD,1 Lyon, France
Formation of a fistula between a retroesophageal subclavian artery and the esophagus is a rare cause of hematemesis that is usually fatal. Several etiologies have been described. The purpose of this report is to describe a case involving successful surgical repair of an arterioesophageal fistula induced by prolonged nasogastric intubation. A preoperative CT scan under emergency conditions allowed tentative diagnosis. Arteriography in the operating room confirmed the presence of a fistula and also allowed temporary hemostasis by tamponade. On the basis of a review of the literature, this case demonstrates the importance of screening patients requiring prolonged nasogastric intubation to rule out the possibility of an aberrant aortic arch system.
Arterioesophageal fistula constitutes a rare, usually fatal event. Some cases develop in the course of esophageal or thoracic disease (neoplasm or infection).1,2 Another mechanism frequently observed in children is perforation of the esophagus after foreign-body ingestion.3 The thoracic aorta is usually involved. The presence of a retroesophageal subclavian artery (RESCA) is also a risk factor for rupture into the esophagus because the close anatomical relationship between the aberrant artery and the upper respiratory and digestive tract. The incidence of RESCA is estimated at between 0.5
1
Service de Chirurgie Vasculaire, Hoˆpital Edouard Herriot, Lyon, France. 2 Service de Urgences Chirurgicales Digestives et Proctologiques, Hoˆpital Edouard Herriot, Lyon, France. 3 Departement d¢Anesthe´sie-Re´animation, Hoˆpital Edouard Herriot, Lyon, France. Correspondence to: P. Feugier, MD, Service de Chirurgie Vasculaire, Pavilion M1, Hoˆpital E Herriot, Place Arsonval, 69437 Lyon, France, E-mail: [email protected] Ann Vasc Surg 2003; 17: 302-305 DOI: 10.1007/s10016-001-0406-z Annals of Vascular Surgery Inc. Published online: 28 April 2003 302
and 1.8% but the exact risk of fistulous complications is difficult to evaluate.4,5 Predisposing factors include aneurysm of the RESCA and prolonged nasogastric or tracheal intubation. The purpose of this report is to describe a case of fistula between the esophagus and a right-sided RESCA in an intensive care unit (ICU) patient and to discuss diagnostic and therapeutic pitfalls based on a review of the literature.
CASE REPORT A 24-year-old man under treatment for chronic alcohol abuse was admitted to intensive care following a defenestration out of his house on fire. He presented with multiple injuries including severe burns over 55% of the body, fracture/compression of vertebrae, and symmetrical pelvic fracture. In addition to intensive care, the patient required several surgical repair procedures, tracheotomy, and feeding via a nasogastric tube. On the 31st day in the ICU, massive hematemesis occurred, leading rapidly to shock. All attempts to perform esophagoscopy were thwarted by the volume of bleeding. After instituting blood pressure support, a
Vol. 17, No. 3, 2003
Fig. 1. CT scan showing a hematoma located in front of the right retroesophageal subclavian artery which is in contact with the nasogastric tube. cervicothoracic computed tomography (CT) scan was performed. Findings demonstrated the presence of a RESCA in contact with the nasogastric tube. Arterioesophageal fistula was suspected because of the presence of hemomediastinum around the cervical esophagus (Fig. 1). After transferring the patient to the operating room, arteriography was undertaken via the right brachial artery. Findings confirmed arterial rupture. Temporary hemostasis was obtained by endoluminal occlusion of the RESCA proximal to the fistula using a catheter 7 mm in diameter. Hematemesis stopped immediately. Open surgical treatment consisted of left anterolateral thoracotomy to perform ligation of the RESCA at its origin, followed by supraclavicular exposure to carry out revascularization of the upper extremity. After 4 days of intensive care with hemodynamic support, the patient underwent esophagectomy with feeding jejunostomy because of the high risk of mediastinitis and the need to resume feeding promptly. Postoperative recovery was good. Respiratory assistance and critical care were gradually discontinued. Antimicrobial treatment was continued. CT scan and ultrasound demonstrated adequate revascularization of the right upper extremity with no neurological or ischemic sequels. After 2 months of intensive care, the patient was discharged from the hospital and sent to a rehabilitation center. He is now able to sit using a support belt. Another procedure is to be undertaken to reestablish digestive continuity.
DISCUSSION Retroesophageal subclavian artery is the most common anomaly of the aortic arch system. Its incidence in the general population is 1/200 indi-
Case reports 303
viduals. It is due to interruption of the fourth right aortic arch between the notches for the common carotid artery and subclavian artery while the left fourth arch remains intact. This anomaly is more frequent in women and mongoloid children.6,7 Since RESCA usually do not produce symptoms, most discoveries have been coincidental. However, dysphagia due to compression of the esophagus is a possible manifestation.8 More rarely, RESCA may be the site of formation of atherosclerotic plaque, inflammatory lesions, or aneurysm. The seriousness of RESCA aneurysm is associated with the high risk of clot-related events, tracheal or venous compression, and rupture.9,10 Formation of a fistula between a nonaneurysmal RESCA and the esophagus is an exceptionally rare event. Our perusal of the literature turned up only 11 cases (Table I). All cases involved young individuals requiring long-term intensive care. Hematemesis was precipitated by esogastric intubation in all cases. The duration of intubation ranged from 9 to 60 days. Fistulization was associated with predisposing factors in three reports—i.e., long-term corticotherapy in two cases, recent surgery of the ascending aorta in two cases, and septicemia in one case. Hemorrhage was fatal in seven cases. The mechanism underlying the development of a fistula involves the induction of limited necrosis of the digestive and arterial walls by pulsatile compression of the esophageal wall between the RESCA and rigid intubation catheter. This leads to thrombosis of the vasa vasorum as well as to ischemia of the digestive wall.22 With time, ulceration and ultimately arteriodigestive fistula appear. This mechanism is probably enhanced by local infectious processes occurring after the esophageal mucosa has been damaged.23 It is also probable that any factor affecting tissue quality or esophageal integrity would act as a contributing factor for fistulization, e.g., prolonged corticotherapy and mediastinitis.15,16,18 Trauma related to deceleration syndrome may have been implicated in our patient but esophagectomy demonstrated no evidence of subadvential rupture. Clinical diagnosis of this complication is difficult because of its extreme rarity. In the absence of any evidence suggesting the possibility of an anomaly of the aortic arch system, there are no specific manifestations. Although two authors described cases involving moderate sentinel hemorrhage heralding hematemesis.15,24 This sign has not been mentioned in most cases and was not present in our patient. The main diagnostic criterion in intensive care patients is occurrence of abrupt, mas-
Recovered 31
Fig. 2. Photographic taken during esophagectomy. The arterioesophageal fistula is 8 mm in diameter with necrosis reaching to 30 mm around the lesion.
M 24
ENT, ear-nose-throat; ETT, endotracheal tube; NGT, nasogastric tube; NM, not mentioned.
M F F F M M F F 27 36 72 0.4 9 55 11 39
Multiple trauma
ETT/NGT
Deceleration syndrome
Fatal Fatal Fatal Recovered Fatal Recovered Recovered NM 60 27 30 NM NM 44 17 NM — Corticotherapy Corticotherapy, infection, surgery — Surgery Surgery, irradiation, cancer — —
M M 25 6
ENT cancer Subarachnoid hemorrhage Recovery after aortic repair Recovery after heart surgery Recovery after heart surgery Esophageal cancer Intracerebral hemorrhage Head trauma
NGT NGT ETT/NGT NGT ETT/NGT NGT ETT/NGT NGT
Fatal Fatal 13 42 — — ETT/NGT ETT/NGT
Fatal F 17
Recovery after cesarian section Head trauma Multiple trauma
NGT
—
9
Annals of Vascular Surgery
Merchant et al., 197711 Livesay et al., 198212 Jungck and Puschel, 198313 Belkin et al., 198414 Edwards et al., 198415 Gossot et al., 198516 Guzzeta et al., 198917 Ikeda et al., 199118 Hirakata et al., 199119 Miller et al., 199620 Minyard and Smith, 200021 Current study
Predisposing factors Cause Pathology requiring intensive care Sex Age (years) Study
Table I. Clinical cases of fistula between a nonaneurysmal RESCA and the esophagus reported in the literature
Time lag after intubation (days)
Outcome
304 Case reports
sive arterial bleeding several days after placement of an endotracheal or nasogastric tube. Esophagoscopy cannot be used to assist diagnosis because of the volume of hematemesis. Perdue et al.25 stated that the sensitivity of endoscopy is low. It has allowed location of aortoesophageal fistula in only two cases.26,27 In addition to delaying surgical management, use of esophagoscopy carries the risk of increasing hemorrhage by removing the hemostatic clot. Similarly, esophagography and magnetic resonance imaging (MRI) are of no use in such emergency situations. In our opinion, cervicothoracic CT scanning with contrast injection is the modality of choice for visualization of RESCA, provided that blood pressure status can be controlled. Cervicothoracic CT scanning also provides strong evidence for arterioesophageal fistula. In the operating room, arteriography via the right brachial artery allowed not only confirmation of diagnosis and location of the site of bleeding but also hemostasis using an occlusion balloon catheter. In our patient, left thoracotomy allowed satisfactory control of the aorta and ligation of the origin of the RESCA that was slightly dilated. Revascularization of the distal RESCA is necessary to avoid severe ischemia of the right upper extremity and vertebrobasilar manifestations.28-30 This was accomplished by transposing the subclavian artery into the carotid artery. In the absence of any sign of mediastinitis, conservative treatment of the esophagus might have been possible in our patient. However, the decision to perform esophagectomy was made because of poor general status as well as the presence of hemomediastinum and bilateral pneumonia involving pyocyanic organisms and Candida albicans (Fig. 2). The extremely unfavorable prognosis of this rare complication combined with the relatively high
Vol. 17, No. 3, 2003
Case reports 305
incidence of RESCA in the general population underlines the need for prevention. If RESCA is diagnosed during aortic arch repair, corrective surgery should be considered. Intensive care patients should be screened before long-term placement of nasogastric tube. Since transesophageal ultrasound is usually diagnostic, a CT scan or magnetic nuclear resonance imaging is usually not required for screening. If a RESCA is diagnosed, the nasogastric tube should be withdrawn and a gastrostomy performed if further feeding is required. Surgical treatment of nonaneurysmal RESCA is unnecessary for patients not presenting with dysphagia.
18.
REFERENCES
19.
1. Marcelo FBJ. Aorto-esophageal fistula: an unusual cause of massive hematemesis and melena. Acta Med Philippina 1965;2:27-34. 2. Taquino GF, Joseph GF. Carcinoma of the oesophagus: an analysis of 145 cases with special reference to metastases and extensions. Ann Otol 1947;56:1041-1058. 3. El Barbary AS, Foad H, Fathi A. Oesophageal fistulae caused by swallowed foreign bodies. J Laryngol Otol 1969;83:251259. 4. Richardson J, Doty D, Rossi N, et al. Operation for aortic arch anomalies. Ann Thorac Surg 1981;31:426-432. 5. Jebara VA, Arnaud-Crozat E, Angel F, et al. Aberrant right subclavian artery aneurysm: report of a case and review of the literature. Ann Thorac Surg 1972;13:559-563. 6. Easterbrook JS. Identification of aberrant right subclavian artery on MR images of the cervical spine. J Magn Reson Imaging 1992;2:507-509. 7. Goldstein WB. Aberrant subclavian artery in mongolism. AJR Am J Roentgenol 1965;93:131-134. 8. Janssen M, Baggen MG, Veen HF, et al. Dysphagia lusoria: clinical aspects, manometric findings, diagnosis, and therapy. Am J Gastroenterol 2000;95:1411-1416. 9. Roland CF, Cherry KJ. Symptomatic atherosclerotic stenotic disease of an aberrant right subclavian artery. Ann Vasc Surg 1991;5:196-198. 10. Schmidt FE, Hewitt RL, Flores Jr AA. Aneurysm of anomalous right subclavian artery. J Vasc Surg 1992;16:90-95. 11. Merchant FJ, Nichols RL, Bombeck CT. Unusual complication of nasogastric esophageal intubation-erosion into an aberrant right subclavian artery. J Cardiovasc Surg 1977;18: 147-150. 12. Livesay JJ, Michals AA, Dainko EC. Anomalous right subclavian arterial esophageal fistula: an unusual complication of tracheostomy. Tex Heart Inst J 1982;9:105-108. 13. Jungck E, Puschel K. Erosion hemorrhage from an esophago-aortic fistula in congenital anomaly of the thoracic
14.
15.
16.
17.
20.
21.
22.
23.
24. 25.
26.
27.
28.
29.
30.
aorta as a fatal complication of a stomach tube. Anaesthesist 1983;32:498-500. Belkin RI, Keller FS, Everts EC, et al. Aberrant right subclavian artery–esophageal fistula: a cause of overwhelming upper gastrointestinal hemorrhage. Cardiovasc Intervent Radiol 1984;7:87-89. Edwards BS, Edwards WD, Connolly DC, et al. Arterialesophageal fistulae developing in patients with anomalies of the aortic arch system. Chest 1984;86:732-735. Gossot D, Nussaume O, Kitzis M, et al. He´ mate´ me`se foudroyante duea I’e´ rosion d’une arte`re sous-clavie`re droite re´ tro-oesophagienne par une sonde oeso-gastrique. Presse Med 1985;14:1655-1656. Guzzetta PC, Newman KD, Ceithaml E. Successful management of aberrant subclavian artery–esophageal fistula in an infant. Ann Thorac Surg 1989;47:308-309. Ikeda T, Yokota Y, Ando F, et al. A case of an aberrant subclavian artery–esophageal fistula due to prolonged nasogastric intubation. Kyobu Geka 1991;44:1045-1047. Hirakata R, Hasuo K, Yasumori K, et al. Arterioenteric fistulae: diagnosis and treatment by angiography. Clin Radiol 1991;43:328-330. Miller RG, Robie DK, Davis SL, et al. Survival after aberrant right subclavian artery–esophageal fistula: case report and literature review. J Vasc Surg 1996;24:271-275. Minyard AN, Smith DM. Arterial-esophageal fistulae in patients requiring nasogastric esophageal intubation. Am J Forensic Med Pathol 2000;21:74-78. Dores GM, Miller ME, Kaufman DG. A herald bleed: a case of aortoesophageal fistula and a review of the literature. R I Med J 1991;74:123-126. Gable DS, Stoddard LD. Acute bacterial aortitis resulting in an aortoesophageal fistula: a fatal complication of untreated esophageal carcinoma. Pathol Res Pract 1989;184:318324. Carter R, Mulder GA, Snyder EN, et al. Aorto-esophageal fistula. Am J Surg 1978;136:26-30. Perdue GD, Smith RB, Aynsley JD, et al. Impending aortoenteric hemorrhage: the effect of early recognition on improved outcome. Ann Surg 1980;192:237-243. Sosnowik D, Greenberg R, Bank S, et al. Aortoesophageal fistula: early and late endoscopic findings. Am J Gastroenterol 1988;83:1401-1404. Ramos MA, Nord HJ. Carotid-esophageal fistula treated with balloon tamponade and surgery. J Vasc Surg 1997;26:144149. Weinberger G, Randall PA, Parker FB, et al. Involvement of an aberrant right subclavian artery in dissection of the thoracic aorta. AJR AM J Roentgenol 1977;129:653-655. Akers Jr DL, Fowl RJ, Plettner J, et al. Complications of anomalous origin of the right subclavian artery: case report and review of the literature. Ann Thorac Surg 1987;44:86-89. Esposito RA, Khalil I, Galloway AC, et al. Surgical treatment for aneurysm of aberrant subclavian artery based on a case report and a review of the literature. Ann Vasc Surg 1991;5:385-388.
Formation of a fistula between a retroesophageal subclavian artery and the esophagus is a rare cause of hematemesis that is usually fatal. Several etiologies have been described. The purpose of this report is to describe a case involving successful surgical repair of an arterioesophageal fistula induced by prolonged nasogastric intubation. A preoperative CT scan under emergency conditions allowed tentative diagnosis. Arteriography in the operating room confirmed the presence of a fistula and also allowed temporary hemostasis by tamponade. On the basis of a review of the literature, this case demonstrates the importance of screening patients requiring prolonged nasogastric intubation to rule out the possibility of an aberrant aortic arch system.
Arterioesophageal fistula constitutes a rare, usually fatal event. Some cases develop in the course of esophageal or thoracic disease (neoplasm or infection).1,2 Another mechanism frequently observed in children is perforation of the esophagus after foreign-body ingestion.3 The thoracic aorta is usually involved. The presence of a retroesophageal subclavian artery (RESCA) is also a risk factor for rupture into the esophagus because the close anatomical relationship between the aberrant artery and the upper respiratory and digestive tract. The incidence of RESCA is estimated at between 0.5
1
Service de Chirurgie Vasculaire, Hoˆpital Edouard Herriot, Lyon, France. 2 Service de Urgences Chirurgicales Digestives et Proctologiques, Hoˆpital Edouard Herriot, Lyon, France. 3 Departement d¢Anesthe´sie-Re´animation, Hoˆpital Edouard Herriot, Lyon, France. Correspondence to: P. Feugier, MD, Service de Chirurgie Vasculaire, Pavilion M1, Hoˆpital E Herriot, Place Arsonval, 69437 Lyon, France, E-mail: [email protected] Ann Vasc Surg 2003; 17: 302-305 DOI: 10.1007/s10016-001-0406-z Annals of Vascular Surgery Inc. Published online: 28 April 2003 302
and 1.8% but the exact risk of fistulous complications is difficult to evaluate.4,5 Predisposing factors include aneurysm of the RESCA and prolonged nasogastric or tracheal intubation. The purpose of this report is to describe a case of fistula between the esophagus and a right-sided RESCA in an intensive care unit (ICU) patient and to discuss diagnostic and therapeutic pitfalls based on a review of the literature.
CASE REPORT A 24-year-old man under treatment for chronic alcohol abuse was admitted to intensive care following a defenestration out of his house on fire. He presented with multiple injuries including severe burns over 55% of the body, fracture/compression of vertebrae, and symmetrical pelvic fracture. In addition to intensive care, the patient required several surgical repair procedures, tracheotomy, and feeding via a nasogastric tube. On the 31st day in the ICU, massive hematemesis occurred, leading rapidly to shock. All attempts to perform esophagoscopy were thwarted by the volume of bleeding. After instituting blood pressure support, a
Vol. 17, No. 3, 2003
Fig. 1. CT scan showing a hematoma located in front of the right retroesophageal subclavian artery which is in contact with the nasogastric tube. cervicothoracic computed tomography (CT) scan was performed. Findings demonstrated the presence of a RESCA in contact with the nasogastric tube. Arterioesophageal fistula was suspected because of the presence of hemomediastinum around the cervical esophagus (Fig. 1). After transferring the patient to the operating room, arteriography was undertaken via the right brachial artery. Findings confirmed arterial rupture. Temporary hemostasis was obtained by endoluminal occlusion of the RESCA proximal to the fistula using a catheter 7 mm in diameter. Hematemesis stopped immediately. Open surgical treatment consisted of left anterolateral thoracotomy to perform ligation of the RESCA at its origin, followed by supraclavicular exposure to carry out revascularization of the upper extremity. After 4 days of intensive care with hemodynamic support, the patient underwent esophagectomy with feeding jejunostomy because of the high risk of mediastinitis and the need to resume feeding promptly. Postoperative recovery was good. Respiratory assistance and critical care were gradually discontinued. Antimicrobial treatment was continued. CT scan and ultrasound demonstrated adequate revascularization of the right upper extremity with no neurological or ischemic sequels. After 2 months of intensive care, the patient was discharged from the hospital and sent to a rehabilitation center. He is now able to sit using a support belt. Another procedure is to be undertaken to reestablish digestive continuity.
DISCUSSION Retroesophageal subclavian artery is the most common anomaly of the aortic arch system. Its incidence in the general population is 1/200 indi-
Case reports 303
viduals. It is due to interruption of the fourth right aortic arch between the notches for the common carotid artery and subclavian artery while the left fourth arch remains intact. This anomaly is more frequent in women and mongoloid children.6,7 Since RESCA usually do not produce symptoms, most discoveries have been coincidental. However, dysphagia due to compression of the esophagus is a possible manifestation.8 More rarely, RESCA may be the site of formation of atherosclerotic plaque, inflammatory lesions, or aneurysm. The seriousness of RESCA aneurysm is associated with the high risk of clot-related events, tracheal or venous compression, and rupture.9,10 Formation of a fistula between a nonaneurysmal RESCA and the esophagus is an exceptionally rare event. Our perusal of the literature turned up only 11 cases (Table I). All cases involved young individuals requiring long-term intensive care. Hematemesis was precipitated by esogastric intubation in all cases. The duration of intubation ranged from 9 to 60 days. Fistulization was associated with predisposing factors in three reports—i.e., long-term corticotherapy in two cases, recent surgery of the ascending aorta in two cases, and septicemia in one case. Hemorrhage was fatal in seven cases. The mechanism underlying the development of a fistula involves the induction of limited necrosis of the digestive and arterial walls by pulsatile compression of the esophageal wall between the RESCA and rigid intubation catheter. This leads to thrombosis of the vasa vasorum as well as to ischemia of the digestive wall.22 With time, ulceration and ultimately arteriodigestive fistula appear. This mechanism is probably enhanced by local infectious processes occurring after the esophageal mucosa has been damaged.23 It is also probable that any factor affecting tissue quality or esophageal integrity would act as a contributing factor for fistulization, e.g., prolonged corticotherapy and mediastinitis.15,16,18 Trauma related to deceleration syndrome may have been implicated in our patient but esophagectomy demonstrated no evidence of subadvential rupture. Clinical diagnosis of this complication is difficult because of its extreme rarity. In the absence of any evidence suggesting the possibility of an anomaly of the aortic arch system, there are no specific manifestations. Although two authors described cases involving moderate sentinel hemorrhage heralding hematemesis.15,24 This sign has not been mentioned in most cases and was not present in our patient. The main diagnostic criterion in intensive care patients is occurrence of abrupt, mas-
Recovered 31
Fig. 2. Photographic taken during esophagectomy. The arterioesophageal fistula is 8 mm in diameter with necrosis reaching to 30 mm around the lesion.
M 24
ENT, ear-nose-throat; ETT, endotracheal tube; NGT, nasogastric tube; NM, not mentioned.
M F F F M M F F 27 36 72 0.4 9 55 11 39
Multiple trauma
ETT/NGT
Deceleration syndrome
Fatal Fatal Fatal Recovered Fatal Recovered Recovered NM 60 27 30 NM NM 44 17 NM — Corticotherapy Corticotherapy, infection, surgery — Surgery Surgery, irradiation, cancer — —
M M 25 6
ENT cancer Subarachnoid hemorrhage Recovery after aortic repair Recovery after heart surgery Recovery after heart surgery Esophageal cancer Intracerebral hemorrhage Head trauma
NGT NGT ETT/NGT NGT ETT/NGT NGT ETT/NGT NGT
Fatal Fatal 13 42 — — ETT/NGT ETT/NGT
Fatal F 17
Recovery after cesarian section Head trauma Multiple trauma
NGT
—
9
Annals of Vascular Surgery
Merchant et al., 197711 Livesay et al., 198212 Jungck and Puschel, 198313 Belkin et al., 198414 Edwards et al., 198415 Gossot et al., 198516 Guzzeta et al., 198917 Ikeda et al., 199118 Hirakata et al., 199119 Miller et al., 199620 Minyard and Smith, 200021 Current study
Predisposing factors Cause Pathology requiring intensive care Sex Age (years) Study
Table I. Clinical cases of fistula between a nonaneurysmal RESCA and the esophagus reported in the literature
Time lag after intubation (days)
Outcome
304 Case reports
sive arterial bleeding several days after placement of an endotracheal or nasogastric tube. Esophagoscopy cannot be used to assist diagnosis because of the volume of hematemesis. Perdue et al.25 stated that the sensitivity of endoscopy is low. It has allowed location of aortoesophageal fistula in only two cases.26,27 In addition to delaying surgical management, use of esophagoscopy carries the risk of increasing hemorrhage by removing the hemostatic clot. Similarly, esophagography and magnetic resonance imaging (MRI) are of no use in such emergency situations. In our opinion, cervicothoracic CT scanning with contrast injection is the modality of choice for visualization of RESCA, provided that blood pressure status can be controlled. Cervicothoracic CT scanning also provides strong evidence for arterioesophageal fistula. In the operating room, arteriography via the right brachial artery allowed not only confirmation of diagnosis and location of the site of bleeding but also hemostasis using an occlusion balloon catheter. In our patient, left thoracotomy allowed satisfactory control of the aorta and ligation of the origin of the RESCA that was slightly dilated. Revascularization of the distal RESCA is necessary to avoid severe ischemia of the right upper extremity and vertebrobasilar manifestations.28-30 This was accomplished by transposing the subclavian artery into the carotid artery. In the absence of any sign of mediastinitis, conservative treatment of the esophagus might have been possible in our patient. However, the decision to perform esophagectomy was made because of poor general status as well as the presence of hemomediastinum and bilateral pneumonia involving pyocyanic organisms and Candida albicans (Fig. 2). The extremely unfavorable prognosis of this rare complication combined with the relatively high
Vol. 17, No. 3, 2003
Case reports 305
incidence of RESCA in the general population underlines the need for prevention. If RESCA is diagnosed during aortic arch repair, corrective surgery should be considered. Intensive care patients should be screened before long-term placement of nasogastric tube. Since transesophageal ultrasound is usually diagnostic, a CT scan or magnetic nuclear resonance imaging is usually not required for screening. If a RESCA is diagnosed, the nasogastric tube should be withdrawn and a gastrostomy performed if further feeding is required. Surgical treatment of nonaneurysmal RESCA is unnecessary for patients not presenting with dysphagia.
18.
REFERENCES
19.
1. Marcelo FBJ. Aorto-esophageal fistula: an unusual cause of massive hematemesis and melena. Acta Med Philippina 1965;2:27-34. 2. Taquino GF, Joseph GF. Carcinoma of the oesophagus: an analysis of 145 cases with special reference to metastases and extensions. Ann Otol 1947;56:1041-1058. 3. El Barbary AS, Foad H, Fathi A. Oesophageal fistulae caused by swallowed foreign bodies. J Laryngol Otol 1969;83:251259. 4. Richardson J, Doty D, Rossi N, et al. Operation for aortic arch anomalies. Ann Thorac Surg 1981;31:426-432. 5. Jebara VA, Arnaud-Crozat E, Angel F, et al. Aberrant right subclavian artery aneurysm: report of a case and review of the literature. Ann Thorac Surg 1972;13:559-563. 6. Easterbrook JS. Identification of aberrant right subclavian artery on MR images of the cervical spine. J Magn Reson Imaging 1992;2:507-509. 7. Goldstein WB. Aberrant subclavian artery in mongolism. AJR Am J Roentgenol 1965;93:131-134. 8. Janssen M, Baggen MG, Veen HF, et al. Dysphagia lusoria: clinical aspects, manometric findings, diagnosis, and therapy. Am J Gastroenterol 2000;95:1411-1416. 9. Roland CF, Cherry KJ. Symptomatic atherosclerotic stenotic disease of an aberrant right subclavian artery. Ann Vasc Surg 1991;5:196-198. 10. Schmidt FE, Hewitt RL, Flores Jr AA. Aneurysm of anomalous right subclavian artery. J Vasc Surg 1992;16:90-95. 11. Merchant FJ, Nichols RL, Bombeck CT. Unusual complication of nasogastric esophageal intubation-erosion into an aberrant right subclavian artery. J Cardiovasc Surg 1977;18: 147-150. 12. Livesay JJ, Michals AA, Dainko EC. Anomalous right subclavian arterial esophageal fistula: an unusual complication of tracheostomy. Tex Heart Inst J 1982;9:105-108. 13. Jungck E, Puschel K. Erosion hemorrhage from an esophago-aortic fistula in congenital anomaly of the thoracic
14.
15.
16.
17.
20.
21.
22.
23.
24. 25.
26.
27.
28.
29.
30.
aorta as a fatal complication of a stomach tube. Anaesthesist 1983;32:498-500. Belkin RI, Keller FS, Everts EC, et al. Aberrant right subclavian artery–esophageal fistula: a cause of overwhelming upper gastrointestinal hemorrhage. Cardiovasc Intervent Radiol 1984;7:87-89. Edwards BS, Edwards WD, Connolly DC, et al. Arterialesophageal fistulae developing in patients with anomalies of the aortic arch system. Chest 1984;86:732-735. Gossot D, Nussaume O, Kitzis M, et al. He´ mate´ me`se foudroyante duea I’e´ rosion d’une arte`re sous-clavie`re droite re´ tro-oesophagienne par une sonde oeso-gastrique. Presse Med 1985;14:1655-1656. Guzzetta PC, Newman KD, Ceithaml E. Successful management of aberrant subclavian artery–esophageal fistula in an infant. Ann Thorac Surg 1989;47:308-309. Ikeda T, Yokota Y, Ando F, et al. A case of an aberrant subclavian artery–esophageal fistula due to prolonged nasogastric intubation. Kyobu Geka 1991;44:1045-1047. Hirakata R, Hasuo K, Yasumori K, et al. Arterioenteric fistulae: diagnosis and treatment by angiography. Clin Radiol 1991;43:328-330. Miller RG, Robie DK, Davis SL, et al. Survival after aberrant right subclavian artery–esophageal fistula: case report and literature review. J Vasc Surg 1996;24:271-275. Minyard AN, Smith DM. Arterial-esophageal fistulae in patients requiring nasogastric esophageal intubation. Am J Forensic Med Pathol 2000;21:74-78. Dores GM, Miller ME, Kaufman DG. A herald bleed: a case of aortoesophageal fistula and a review of the literature. R I Med J 1991;74:123-126. Gable DS, Stoddard LD. Acute bacterial aortitis resulting in an aortoesophageal fistula: a fatal complication of untreated esophageal carcinoma. Pathol Res Pract 1989;184:318324. Carter R, Mulder GA, Snyder EN, et al. Aorto-esophageal fistula. Am J Surg 1978;136:26-30. Perdue GD, Smith RB, Aynsley JD, et al. Impending aortoenteric hemorrhage: the effect of early recognition on improved outcome. Ann Surg 1980;192:237-243. Sosnowik D, Greenberg R, Bank S, et al. Aortoesophageal fistula: early and late endoscopic findings. Am J Gastroenterol 1988;83:1401-1404. Ramos MA, Nord HJ. Carotid-esophageal fistula treated with balloon tamponade and surgery. J Vasc Surg 1997;26:144149. Weinberger G, Randall PA, Parker FB, et al. Involvement of an aberrant right subclavian artery in dissection of the thoracic aorta. AJR AM J Roentgenol 1977;129:653-655. Akers Jr DL, Fowl RJ, Plettner J, et al. Complications of anomalous origin of the right subclavian artery: case report and review of the literature. Ann Thorac Surg 1987;44:86-89. Esposito RA, Khalil I, Galloway AC, et al. Surgical treatment for aneurysm of aberrant subclavian artery based on a case report and a review of the literature. Ann Vasc Surg 1991;5:385-388.