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Spontaneous Dissection of the Celiac Artery
Case Reports
Spontaneous Dissection of the Celiac Artery Philippe Chaillou, Philippe Moussu, Sarah-Frederique Noel, Christine Sagan, Marc-Antoine Pistorius, Jean-Marc Langlard, and Philippe Patra, Nantes, France
We describe a case of intestinal angina caused by spontaneous dissection of the celiac artery and thrombosis of the superior mesenteric artery. Spontaneous dissection of a visceral artery is an uncommon occurrence that is usually diagnosed after fatal hemorrhage or ischemia. The underlying mechanism is unclear but the frequent association with multiple arterial lesions suggests general arterial disease. In symptomatic forms, surgical reconstruction is mandatory, to treat the lesion and allow definitive histological diagnosis. (Ann Vasc Surg 1997;11:413-415.)
CASE REPORT In April 1993, a 64-year-old woman consulted for postprandial abdominal pain that led to a loss of 11 kg in 2 months by limiting food intake. The patient had suffered infero-latero-basal myocardial infarction, complicated by paroxysmal atrial fibrillation, and hemispheric transient ischemic attack in 1989, and thrombosis of the central retinal artery in 1990. Polycythemia vera was diagnosed in January 1993 and successfully controlled by drug therapy (Vercyte, 0.5 mg/kg). There were no other risk factors for cardiovascular disease such as smoking, hormone therapy, or dyslipemia. Clinical examination revealed only epigastric murmur. The diagnosis of intestinal angina was proposed after ruling out gastroduodenal peptic disease or pancreatic disease. Doppler ultrasound and abdominal CT-scan revealed thrombosis and calcification of the superior mesenteric artery (SMA). Angiography demonstrated proximal occlusion of the celiac artery (CA) and SMA. The distal celiac and superior mesenteric arteries were supplied retrogradely from the inferior mesenteric artery via a highly developed Riolan’s arch. The aorta and renal arteries appeared normal with no sign of atheroma or dysplasia (Figs. 1 and 2). Median xipho-pubic transperitoneal laparotomy was
From the Clinique Chirurgicale Thoracique, Cardiaque et Vasculaire, Laboratoire d’Anatomo-pathologie and Clinique Cardiologique, Hopital Rene´ et Guillaume Laennec, and the Service de Medecine Interne, Hotel Dieu, Nantes, France. Correspondence to: P. Chaillou, MD, Clinique Chirurgicale Thoracique, Cardiaque et Vasculaire, Hopital Guillaume et Rene´ Laennec, 44035 Nantes, France.
performed. Intraoperative examination showed that the CA was occluded from the ostium to the origin of the collaterals, which were patent. An embolic occlusion was also present in the SMA distal to the first branches. The macroscopic appearance was not suggestive of dissection. Bypass was achieved by placement of two PTFE prosthetic grafts (Gore-tex™) on the anterior aspect of the supraceliac aorta with end-to-end anastomosis on the CA and SMA via an anterograde route. Postoperative recovery was uneventful with resumption of normal alimentation. The patient was discharged on postoperative day 10. Histologic examination demonstrated dissection of the CA (Fig. 3). The cleavage plane was located in the external elastic layer which appeared to be doubled. Both the false and true lumens were thrombosed. The intimal tear could not be located. The SMA was the site of previous organized thrombosis without characteristic evidence. Six months postoperatively, the patient was in good health and ultrasound imaging showed patent bypasses.
DISCUSSION Arterial dissection is defined as cleavage of the wall by an intramural hematoma located between two elastic layers.1 Unlike aortic dissection which is relatively common,2 spontaneous dissection of peripheral arteries unrelated to the aorta are rare. Cases reported in the literature have involved the renal arteries, the coronary arteries, intracranial arteries, and visceral arteries, in order of incidence.1 In visceral arteries, the most frequent site is the SMA (16 cases compiled by Krupski et al. in 1985).3 413
414
Case reports
Annals of Vascular Surgery
Fig. 1. Aortic angiography showing no opacification of the celiac trunk and the superior mesenteric artery as well as a highly developed Riolan’s arch. Fig. 2. Selective arteriography of the inferior mesenteric artery demonstrating hypertrophy of the Riolan’s arch.
Fig. 3. Transverse section of the celiac artery at low magnification (Weigert staining revealing elastic fibers). The cleavage plane is located in the external elastic layer.
Involvement of branches of the CA, the splenic artery,4 the left gastric artery, and especially the hepatic artery is more uncommon; a total of only 13 cases were reported in 1988.5 Diagnosis of isolated dissection of the CA is exceptional. Two cases of
spontaneous dissection of the CA extending to branches have been described in separate reports by Foord and Lewis in 19591 and Bartoli et al. in 1990.6 About half of all dissections involving visceral arteries are asymptomatic. Diagnosis is usually made at autopsy.1,4,5,7 Occasionally arterial dissection is revealed by intestinal angina. Our patient presented the three cardinal signs of intestinal angina: pain, weight loss, and epigastric murmur.8 However, more atypical symptoms have been described depending on the location of the arterial lesions, e.g., malabsorption in association with dissection of the SMA,9 jaundice in association with dissection of the hepatic artery10 or pancreatitis with elevated serum amylase levels.11 Most often dissection is revealed by acute ischemia (intestinal infarction) 1,12,–14 or hemorrhage (hemoperitoneum, retroperitoneal hematoma).5,6,11,15–18 These acute manifestations are usually fatal (13 out of the 15 cases reported). Bowel infarction did not occur in our case because of a highly developed collateral circulation. The fact that the lesions formed progressively in
Vol. 11, No. 4, 1997
two stages probably allowed time for this development. Neither the cause (embolism or polyglobulia) nor age of thrombosis of the SMA could be determined. In most cases imaging techniques suggest tentative diagnosis. Ultrasound study and CT-scan with contrast injection can visualize two separate channels in the dissected wall.5,8 Selective arteriography is the method of choice to confirm diagnosis by demonstrating a double lumen. However, this finding can be masked either completely by arterial occlusion as was the case in our patient, or partially if only one channel is circulating, resulting in an aspect of nonspecific stenosis. Posttraumatic dissection (11 out of 48 in the series reported by Foord and Lewis1) must be distinguished from spontaneous dissection for which no etiology has been proposed. Arterial hypertension, female sex, and pregnancy have been described as predisposing factors.6,11,19 Our patient had not suffered trauma and was not hypertensive. The female risk factor which was based on experience with dissection of the coronary arteries19 has not been confirmed for visceral artery dissections (14 women in the 30 cases reported in the literature). From a histological standpoint, dissection occurs between the intima and the external elastic layer. This is in contrast with aortic dissection in which the cleavage plane is located between the first and second third of the intima.1,17 It is common to observe cystic necrosis of the media6,14,15 but this finding is probably not specific.1 Pinkerton et al.20 described one case of hepatic artery dissection due to fibromuscular dysplasia. Dissected arteries are rarely atherosclerotic and the intimal tear is rarely detected.1 In our case, the CA showed no notable dystrophy. Since multiple arterial dissections are relatively frequent,1 it could be suspected that in our patient a coexisting coronary artery dissection determined the previous myocardial infarction. However coronarography was not performed since there were no clinical symptoms. Reconstruction is necessary in patients with symptomatic intestinal ischemia associated with weight loss. Restoration of circulation in all arteries is advisable if the patient’s condition allows.9 We performed bypass using a PTFE graft for both arteries. Direct transposition into the aorta, the preferred technique for atherosclerotic lesions of the SMA,9 was not feasible in our case because of the length of the thrombosed segment. The choice of graft material (vein, Dacron, PTFE, or arterial autograft) and route of the bypass (anterograde or retrograde) vary
Case reports
415
from one author to another with no obvious difference in the quality of results. Regardless of reconstruction technique used, an arterial biopsy should be taken to confirm diagnosis.9 REFERENCES 1. Foord AG, Lewis RD. Primary dissecting aneurysms of peripheral and pulmonary arteries. Arch Path 1959;68:553577. 2. Ferrier A, Delorme G. Pathologie de l’aorte thoracique. In Godeau P, ed. Traite´ de Me´decine. Paris: Flammarion, 1987, pp 961-966. 3. Krupski WC, Effeney DJ, Ehrenfeld WK. Spontaneous dissection of the superior mesenteric artery. J Vasc Surg 1985; 2:731-734. 4. Watson AJ. Dissecting aneurysm of arteries other than the aorta. J Pathol Bacteriol 1956;72:439-449. 5. Derhy S, Foster DW, Cauquil P, et al. Dissection spontane´e de l’arte`re he´patique: a` propos d’un cas. Ann Radiol 1988; 31:301-304. 6. Bartoli JM, Moulin G, Di Stefano D, et al. Dissection isole´e du tronc caeliaque et de ses branches. Ann Radiol 1990;33: 264-266. 7. Contreras-Guerrero E. Primary dissecting aneurysm of the hepatic artery. Arch Path 1970;89:569-573. 8. Kieny R. et Cinqualbre J. Chirurgie des arte`res digestives. E. M. C. Paris, Techniques chirurgicales, Chirurgie Vasculaire, 4.7. 12, 43105. 9. Clark F, Murray MD. Steatorrhoea due to dissecting aneurysm of the superior mesenteric artery. Br Med J 1962;2: 965-966. 10. Perreau P, Simard C, Bouali A, Bertrand G. Ane´vrysme disse´quant de l’arte`re he´patique. Presse Med 1970;78:19351937. 11. Richard JP, Choquard P, Viellefond A. Dissection isole´e d’une arte`re pancre´atique. Nouv Presse Med 1975;4:271272. 12. Corbetti F, Vigo M, Bulzacchi A, et al. Diagnosis of spontaneous dissection of the superior mesenteric artery. J Comput Assist Tomogr 1989;13:965-967. 13. Bauersfeld SR. Dissecting aneurysm of aorta: presentation of 15 cases and a review of recent literature. Ann Intern Med 1947;26:873-889. 14. Ramchand S, Suh HS, Gonzales-Crussi F. Dissecting aneurysm of the superior mesenteric artery. Can Med Assoc J 1969;101:356-358. 15. Hill DE, Lobell M, Edwards JE. Primary dissecting aneurysm of the hepatic artery. Arch Int Med 1974;133:471-474. 16. Jean C, Marois M, Brochu P. Ane´vrysme disse´quant primitif de l’arte`re me´sente´rique supe´rieure. Can Med Assoc J 1961; 85:942-943. 17. Callicot JH, Hoke HF. Dissecting aneurysm of the common hepatic artery. Arch Path 1968;85:681-685. 18. Cormier JM, Diebold J, Kalifat R, et coll. Rupture arte´rielle spontane´e par dissection de l’arte`re me´sente´rique infe´rieure. Presse Med 1969;77:971-973. 19. Chokron R, Thomas D, Leprince JL, et coll. Dissection primitives isole´es des arte`res coronaires: a` propos de 3 cas d’e´volution favorable. Arch Mal Coeur 1983;76:886-892. 20. Pinkerton JA, Wood WG, Fowler D. Fibrodysplasia with dissecting aneurysm of the hepatic artery. Surgery 1976;79: 721-723.
Spontaneous Dissection of the Celiac Artery Philippe Chaillou, Philippe Moussu, Sarah-Frederique Noel, Christine Sagan, Marc-Antoine Pistorius, Jean-Marc Langlard, and Philippe Patra, Nantes, France
We describe a case of intestinal angina caused by spontaneous dissection of the celiac artery and thrombosis of the superior mesenteric artery. Spontaneous dissection of a visceral artery is an uncommon occurrence that is usually diagnosed after fatal hemorrhage or ischemia. The underlying mechanism is unclear but the frequent association with multiple arterial lesions suggests general arterial disease. In symptomatic forms, surgical reconstruction is mandatory, to treat the lesion and allow definitive histological diagnosis. (Ann Vasc Surg 1997;11:413-415.)
CASE REPORT In April 1993, a 64-year-old woman consulted for postprandial abdominal pain that led to a loss of 11 kg in 2 months by limiting food intake. The patient had suffered infero-latero-basal myocardial infarction, complicated by paroxysmal atrial fibrillation, and hemispheric transient ischemic attack in 1989, and thrombosis of the central retinal artery in 1990. Polycythemia vera was diagnosed in January 1993 and successfully controlled by drug therapy (Vercyte, 0.5 mg/kg). There were no other risk factors for cardiovascular disease such as smoking, hormone therapy, or dyslipemia. Clinical examination revealed only epigastric murmur. The diagnosis of intestinal angina was proposed after ruling out gastroduodenal peptic disease or pancreatic disease. Doppler ultrasound and abdominal CT-scan revealed thrombosis and calcification of the superior mesenteric artery (SMA). Angiography demonstrated proximal occlusion of the celiac artery (CA) and SMA. The distal celiac and superior mesenteric arteries were supplied retrogradely from the inferior mesenteric artery via a highly developed Riolan’s arch. The aorta and renal arteries appeared normal with no sign of atheroma or dysplasia (Figs. 1 and 2). Median xipho-pubic transperitoneal laparotomy was
From the Clinique Chirurgicale Thoracique, Cardiaque et Vasculaire, Laboratoire d’Anatomo-pathologie and Clinique Cardiologique, Hopital Rene´ et Guillaume Laennec, and the Service de Medecine Interne, Hotel Dieu, Nantes, France. Correspondence to: P. Chaillou, MD, Clinique Chirurgicale Thoracique, Cardiaque et Vasculaire, Hopital Guillaume et Rene´ Laennec, 44035 Nantes, France.
performed. Intraoperative examination showed that the CA was occluded from the ostium to the origin of the collaterals, which were patent. An embolic occlusion was also present in the SMA distal to the first branches. The macroscopic appearance was not suggestive of dissection. Bypass was achieved by placement of two PTFE prosthetic grafts (Gore-tex™) on the anterior aspect of the supraceliac aorta with end-to-end anastomosis on the CA and SMA via an anterograde route. Postoperative recovery was uneventful with resumption of normal alimentation. The patient was discharged on postoperative day 10. Histologic examination demonstrated dissection of the CA (Fig. 3). The cleavage plane was located in the external elastic layer which appeared to be doubled. Both the false and true lumens were thrombosed. The intimal tear could not be located. The SMA was the site of previous organized thrombosis without characteristic evidence. Six months postoperatively, the patient was in good health and ultrasound imaging showed patent bypasses.
DISCUSSION Arterial dissection is defined as cleavage of the wall by an intramural hematoma located between two elastic layers.1 Unlike aortic dissection which is relatively common,2 spontaneous dissection of peripheral arteries unrelated to the aorta are rare. Cases reported in the literature have involved the renal arteries, the coronary arteries, intracranial arteries, and visceral arteries, in order of incidence.1 In visceral arteries, the most frequent site is the SMA (16 cases compiled by Krupski et al. in 1985).3 413
414
Case reports
Annals of Vascular Surgery
Fig. 1. Aortic angiography showing no opacification of the celiac trunk and the superior mesenteric artery as well as a highly developed Riolan’s arch. Fig. 2. Selective arteriography of the inferior mesenteric artery demonstrating hypertrophy of the Riolan’s arch.
Fig. 3. Transverse section of the celiac artery at low magnification (Weigert staining revealing elastic fibers). The cleavage plane is located in the external elastic layer.
Involvement of branches of the CA, the splenic artery,4 the left gastric artery, and especially the hepatic artery is more uncommon; a total of only 13 cases were reported in 1988.5 Diagnosis of isolated dissection of the CA is exceptional. Two cases of
spontaneous dissection of the CA extending to branches have been described in separate reports by Foord and Lewis in 19591 and Bartoli et al. in 1990.6 About half of all dissections involving visceral arteries are asymptomatic. Diagnosis is usually made at autopsy.1,4,5,7 Occasionally arterial dissection is revealed by intestinal angina. Our patient presented the three cardinal signs of intestinal angina: pain, weight loss, and epigastric murmur.8 However, more atypical symptoms have been described depending on the location of the arterial lesions, e.g., malabsorption in association with dissection of the SMA,9 jaundice in association with dissection of the hepatic artery10 or pancreatitis with elevated serum amylase levels.11 Most often dissection is revealed by acute ischemia (intestinal infarction) 1,12,–14 or hemorrhage (hemoperitoneum, retroperitoneal hematoma).5,6,11,15–18 These acute manifestations are usually fatal (13 out of the 15 cases reported). Bowel infarction did not occur in our case because of a highly developed collateral circulation. The fact that the lesions formed progressively in
Vol. 11, No. 4, 1997
two stages probably allowed time for this development. Neither the cause (embolism or polyglobulia) nor age of thrombosis of the SMA could be determined. In most cases imaging techniques suggest tentative diagnosis. Ultrasound study and CT-scan with contrast injection can visualize two separate channels in the dissected wall.5,8 Selective arteriography is the method of choice to confirm diagnosis by demonstrating a double lumen. However, this finding can be masked either completely by arterial occlusion as was the case in our patient, or partially if only one channel is circulating, resulting in an aspect of nonspecific stenosis. Posttraumatic dissection (11 out of 48 in the series reported by Foord and Lewis1) must be distinguished from spontaneous dissection for which no etiology has been proposed. Arterial hypertension, female sex, and pregnancy have been described as predisposing factors.6,11,19 Our patient had not suffered trauma and was not hypertensive. The female risk factor which was based on experience with dissection of the coronary arteries19 has not been confirmed for visceral artery dissections (14 women in the 30 cases reported in the literature). From a histological standpoint, dissection occurs between the intima and the external elastic layer. This is in contrast with aortic dissection in which the cleavage plane is located between the first and second third of the intima.1,17 It is common to observe cystic necrosis of the media6,14,15 but this finding is probably not specific.1 Pinkerton et al.20 described one case of hepatic artery dissection due to fibromuscular dysplasia. Dissected arteries are rarely atherosclerotic and the intimal tear is rarely detected.1 In our case, the CA showed no notable dystrophy. Since multiple arterial dissections are relatively frequent,1 it could be suspected that in our patient a coexisting coronary artery dissection determined the previous myocardial infarction. However coronarography was not performed since there were no clinical symptoms. Reconstruction is necessary in patients with symptomatic intestinal ischemia associated with weight loss. Restoration of circulation in all arteries is advisable if the patient’s condition allows.9 We performed bypass using a PTFE graft for both arteries. Direct transposition into the aorta, the preferred technique for atherosclerotic lesions of the SMA,9 was not feasible in our case because of the length of the thrombosed segment. The choice of graft material (vein, Dacron, PTFE, or arterial autograft) and route of the bypass (anterograde or retrograde) vary
Case reports
415
from one author to another with no obvious difference in the quality of results. Regardless of reconstruction technique used, an arterial biopsy should be taken to confirm diagnosis.9 REFERENCES 1. Foord AG, Lewis RD. Primary dissecting aneurysms of peripheral and pulmonary arteries. Arch Path 1959;68:553577. 2. Ferrier A, Delorme G. Pathologie de l’aorte thoracique. In Godeau P, ed. Traite´ de Me´decine. Paris: Flammarion, 1987, pp 961-966. 3. Krupski WC, Effeney DJ, Ehrenfeld WK. Spontaneous dissection of the superior mesenteric artery. J Vasc Surg 1985; 2:731-734. 4. Watson AJ. Dissecting aneurysm of arteries other than the aorta. J Pathol Bacteriol 1956;72:439-449. 5. Derhy S, Foster DW, Cauquil P, et al. Dissection spontane´e de l’arte`re he´patique: a` propos d’un cas. Ann Radiol 1988; 31:301-304. 6. Bartoli JM, Moulin G, Di Stefano D, et al. Dissection isole´e du tronc caeliaque et de ses branches. Ann Radiol 1990;33: 264-266. 7. Contreras-Guerrero E. Primary dissecting aneurysm of the hepatic artery. Arch Path 1970;89:569-573. 8. Kieny R. et Cinqualbre J. Chirurgie des arte`res digestives. E. M. C. Paris, Techniques chirurgicales, Chirurgie Vasculaire, 4.7. 12, 43105. 9. Clark F, Murray MD. Steatorrhoea due to dissecting aneurysm of the superior mesenteric artery. Br Med J 1962;2: 965-966. 10. Perreau P, Simard C, Bouali A, Bertrand G. Ane´vrysme disse´quant de l’arte`re he´patique. Presse Med 1970;78:19351937. 11. Richard JP, Choquard P, Viellefond A. Dissection isole´e d’une arte`re pancre´atique. Nouv Presse Med 1975;4:271272. 12. Corbetti F, Vigo M, Bulzacchi A, et al. Diagnosis of spontaneous dissection of the superior mesenteric artery. J Comput Assist Tomogr 1989;13:965-967. 13. Bauersfeld SR. Dissecting aneurysm of aorta: presentation of 15 cases and a review of recent literature. Ann Intern Med 1947;26:873-889. 14. Ramchand S, Suh HS, Gonzales-Crussi F. Dissecting aneurysm of the superior mesenteric artery. Can Med Assoc J 1969;101:356-358. 15. Hill DE, Lobell M, Edwards JE. Primary dissecting aneurysm of the hepatic artery. Arch Int Med 1974;133:471-474. 16. Jean C, Marois M, Brochu P. Ane´vrysme disse´quant primitif de l’arte`re me´sente´rique supe´rieure. Can Med Assoc J 1961; 85:942-943. 17. Callicot JH, Hoke HF. Dissecting aneurysm of the common hepatic artery. Arch Path 1968;85:681-685. 18. Cormier JM, Diebold J, Kalifat R, et coll. Rupture arte´rielle spontane´e par dissection de l’arte`re me´sente´rique infe´rieure. Presse Med 1969;77:971-973. 19. Chokron R, Thomas D, Leprince JL, et coll. Dissection primitives isole´es des arte`res coronaires: a` propos de 3 cas d’e´volution favorable. Arch Mal Coeur 1983;76:886-892. 20. Pinkerton JA, Wood WG, Fowler D. Fibrodysplasia with dissecting aneurysm of the hepatic artery. Surgery 1976;79: 721-723.