Case Report Ruptured Carotid Aneurysm Revealing a Behcet’s Disease Youssef Lyazidi, Ghislain Y. Abissegue, Hassan T. Chtata, and Mustafa Taberkant, Rabat, Morocco
We report the case of a 20-year-old male who was operated for a large ruptured aneurysm of the right common carotid artery, revealing a Behcet’s disease. The aneurysm was excised and the right common carotid artery was repaired with a polytetrafluoroethylene prosthesis. None of the criteria of the International Study Group for Behcet’s disease was present at the time of the diagnosis.
Carotid aneurysms are characterized by the variety of their etiologies, and this localization is very seldom observed during Behcet’s disease (BD). We report a case of BD revealed by a very large ruptured aneurysm of the right common carotid artery.
CASE REPORT A 20-year-old patient without significant pathological history presented in the emergency room for a right cervical mass (Fig. 1) evolving since 1 month and very painful for 2 days. The clinical examination found a huge pulsatile and expansive right cervical mass with an exquisite pain at palpation. No neurological deficit was noted. The interrogation revealed an oral aphthosis with few recurrences and without genital aphthosis. Biological tests did not find an inflammatory syndrome. The cervical angio-computed tomography objectified a giant 90-mm ruptured aneurysm of the right common carotid artery with a large parietal thrombus, pushing back the trachea and the esophagus (Fig. 2). Because of the type of the arterial lesions and their association with a recurrent
Service de Chirurgie Vasculaire, H^opital Militaire d’Instruction Mohammed V, Rabat, Morocco. Correspondence to: Youssef Lyazidi, MD, Service de Chirurgie Vasculaire, H^opital Militaire d’Instruction Mohammed V, Rabat, Morocco; E-mail: [email protected] Ann Vasc Surg 2015; -: 1–4 http://dx.doi.org/10.1016/j.avsg.2015.02.024 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: October 30, 2014; manuscript accepted: February 6, 2015; published online: ---.
oral aphthosis, the diagnosis of inflammatory carotid aneurysm complicating BD was then retained and our patient was brought into the operating room in emergency. Open aneurysmectomy was performed. Sternotomy allowed the control of the brachiocephalic trunk. It was associated with a presternocleidomastoid approach, and arterial repair was done using a 6-mm polytetrafluoroethylene (PTFE) prosthetic bypass (Fig. 3). Postoperative course was uneventful and the patient left the hospital on the sixth day. The pathological study revealed a nonatheromatous arterial wall, with an important inflammatory medial and adventitial infiltrate. Bacteriological samples did not grow bacteria. A long-term treatment with corticoids, colchicine, and aspirin was initiated. The patency of the carotid bypass was checked 1 month after the intervention by ultrasound.
DISCUSSION Aneurysms of the extracranial carotid arteries are rare, with an annual incidence of 0.5e1% in the surgical literature.1 In the past, before the era of the antibiotic treatment, such aneurysms occurred primarily during infections. Currently, atherosclerosis and post-traumatic, dysplastic, or dissecting aneurysms are the most frequent causes. Postoperative false aneurysms of the carotid bifurcation should also be considered and may appear several years after a carotid endarterectomy with patch angioplasty.2 The occurrence of such aneurysms in BD is very rare. Indeed, the most frequent localizations of 1
2 Case Report
Annals of Vascular Surgery
Fig. 1. Large right mass of the neck revealing a carotid aneurysm. (A) Lateral view. (B) Frontal view.
Fig. 2. Angio-CT objectifying a giant 90-mm ruptured aneurysm of the right common carotid artery. (A) Axial cut showing the exact site of aneurysm in neck. (B) Sagittal cut showing the shape of the aneurysm and its relation to cervical spine.
aneurysms in BD are the aorta, the pulmonary arteries, and the femoral arteries.3 In our patient, the ruptured carotid aneurysm was inaugural and revealed BD. Rupture is an exceptional complication of carotid aneurysms and only some cases were described in the literature.4 Various methods of treatment of the carotid aneurysms were reported in the literature, including open repair with venous or prosthetic bypass, patch angioplasty, and carotid bypass,5 as well as endovascular repair using covered stents or coil embolization.6 The choice of an adequate technique in BD is difficult and still debated. In our patient, we chose the use of a prosthetic PTFE bypass considering the frequency of venous thromboses in BD.7 Late complications are
frequent, in particular anastomotic false aneurysms, observed especially during the active phase of the disease.8 The choice of an anastomotic site in a healthy zone far from the inflammatory edges of the artery and the reinforcement of the sutures with Teflon pledgets might reduce the incidence of the secondary false aneurysms.9 The endovascular treatment of carotid aneurysms using covered stents seems to reduce the incidence of peroperative complications, but it may be complicated by asymptomatic thrombosis of the stent.7 Such an approach could not be tried on our patient because of the giant size of aneurysm and the rupture. A perioperative medical treatment associating corticoids and immunosuppressive drugs is always
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Fig. 3. Treatment of the ruptured aneurysm by sternotomy and a cervical approach with a PTFE bypass graft. (A) Appearance after evacuation of aneurysmal thrombus and section of the arterial defect. (B) Control of brachiocephalic artery. (C) Prosthetic graft bypass repair of carotid artery. (D) patient at 2nd postoperative day.
necessary to control the activity of the disease and it seems to reduce some postoperative complications.10
CONCLUSION The arterial manifestations of the BD are serious but seldom inaugural. In our patient, the rupture of a carotid aneurysm revealed the disease. Surgical aneurysmectomy does not always give good long-term results, and carotid ligation was proposed
by several authors as a simple and definitive alternative method, with less late complications. Endovascular exclusion seems to be a promising option, but studies are necessary to evaluate its long-term advantages.
REFERENCES 1. Davidovic L, Dusan K, Maksimovic Z, et al. Carotid artery aneurysms. Vascular 2004;12:166e70.
4 Case Report
2. Ghilardi G, Masseto N, Cattalini C, et al. Brain involvement in extracranial internal carotid artery aneurysms. Vasa 2001;30:37e41. 3. Sayed A, Elwan H, Fouad F, et al. Behcet extracranial carotid aneurysms: is there still a role for ligation? Eur J Vasc Endovasc Surg 2010;39:17e22. 4. Tuzun H, Besirli K, Sayin A, et al. Management of aneurysms in Behcet syndrome: an analysis of 24 patients. Surgery 1997;121:150e6. 5. Ozyazicioglu A, Kocak H, Vural U. Carotid artery pseudoaneurysm in Behcet disease. Eur J Cardiothorac Surg 2001;19:938e9. 6. Kwon Koo B, Shim WH, Yoon YS, et al. Endovascular therapy combined with immunosuppressive treatment for
Annals of Vascular Surgery
7.
8.
9. 10.
pseudoaneurysms in patients with Behcet disease. J Endovasc Ther 2003;10:75e80. Berard X, Corpataux JM, Taoufiq H, et al. Don’t trust a vein graft to treat carotid aneurysm in patients with Behcet disease. Vasc Surg 2010;52:471e4. Kuzu MA, Ozaslan C, Koksoy C, et al. Vascular involvement in Behcet disease: 8-year audit. World J Surg 1994;18: 948e53. Bradbury AW, Milne AA, Murie JA. Surgical aspects of Behcet disease. Br J Surg 1994;81:1712e21. Yekeler E, Tunaci A, Tunaci M, et al. Successful medical treatment of abdominal aortic aneurysms in a patient with Behcet disease: imaging findings. Australas Radiol 2005;49: 182e4.
We report the case of a 20-year-old male who was operated for a large ruptured aneurysm of the right common carotid artery, revealing a Behcet’s disease. The aneurysm was excised and the right common carotid artery was repaired with a polytetrafluoroethylene prosthesis. None of the criteria of the International Study Group for Behcet’s disease was present at the time of the diagnosis.
Carotid aneurysms are characterized by the variety of their etiologies, and this localization is very seldom observed during Behcet’s disease (BD). We report a case of BD revealed by a very large ruptured aneurysm of the right common carotid artery.
CASE REPORT A 20-year-old patient without significant pathological history presented in the emergency room for a right cervical mass (Fig. 1) evolving since 1 month and very painful for 2 days. The clinical examination found a huge pulsatile and expansive right cervical mass with an exquisite pain at palpation. No neurological deficit was noted. The interrogation revealed an oral aphthosis with few recurrences and without genital aphthosis. Biological tests did not find an inflammatory syndrome. The cervical angio-computed tomography objectified a giant 90-mm ruptured aneurysm of the right common carotid artery with a large parietal thrombus, pushing back the trachea and the esophagus (Fig. 2). Because of the type of the arterial lesions and their association with a recurrent
Service de Chirurgie Vasculaire, H^opital Militaire d’Instruction Mohammed V, Rabat, Morocco. Correspondence to: Youssef Lyazidi, MD, Service de Chirurgie Vasculaire, H^opital Militaire d’Instruction Mohammed V, Rabat, Morocco; E-mail: [email protected] Ann Vasc Surg 2015; -: 1–4 http://dx.doi.org/10.1016/j.avsg.2015.02.024 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: October 30, 2014; manuscript accepted: February 6, 2015; published online: ---.
oral aphthosis, the diagnosis of inflammatory carotid aneurysm complicating BD was then retained and our patient was brought into the operating room in emergency. Open aneurysmectomy was performed. Sternotomy allowed the control of the brachiocephalic trunk. It was associated with a presternocleidomastoid approach, and arterial repair was done using a 6-mm polytetrafluoroethylene (PTFE) prosthetic bypass (Fig. 3). Postoperative course was uneventful and the patient left the hospital on the sixth day. The pathological study revealed a nonatheromatous arterial wall, with an important inflammatory medial and adventitial infiltrate. Bacteriological samples did not grow bacteria. A long-term treatment with corticoids, colchicine, and aspirin was initiated. The patency of the carotid bypass was checked 1 month after the intervention by ultrasound.
DISCUSSION Aneurysms of the extracranial carotid arteries are rare, with an annual incidence of 0.5e1% in the surgical literature.1 In the past, before the era of the antibiotic treatment, such aneurysms occurred primarily during infections. Currently, atherosclerosis and post-traumatic, dysplastic, or dissecting aneurysms are the most frequent causes. Postoperative false aneurysms of the carotid bifurcation should also be considered and may appear several years after a carotid endarterectomy with patch angioplasty.2 The occurrence of such aneurysms in BD is very rare. Indeed, the most frequent localizations of 1
2 Case Report
Annals of Vascular Surgery
Fig. 1. Large right mass of the neck revealing a carotid aneurysm. (A) Lateral view. (B) Frontal view.
Fig. 2. Angio-CT objectifying a giant 90-mm ruptured aneurysm of the right common carotid artery. (A) Axial cut showing the exact site of aneurysm in neck. (B) Sagittal cut showing the shape of the aneurysm and its relation to cervical spine.
aneurysms in BD are the aorta, the pulmonary arteries, and the femoral arteries.3 In our patient, the ruptured carotid aneurysm was inaugural and revealed BD. Rupture is an exceptional complication of carotid aneurysms and only some cases were described in the literature.4 Various methods of treatment of the carotid aneurysms were reported in the literature, including open repair with venous or prosthetic bypass, patch angioplasty, and carotid bypass,5 as well as endovascular repair using covered stents or coil embolization.6 The choice of an adequate technique in BD is difficult and still debated. In our patient, we chose the use of a prosthetic PTFE bypass considering the frequency of venous thromboses in BD.7 Late complications are
frequent, in particular anastomotic false aneurysms, observed especially during the active phase of the disease.8 The choice of an anastomotic site in a healthy zone far from the inflammatory edges of the artery and the reinforcement of the sutures with Teflon pledgets might reduce the incidence of the secondary false aneurysms.9 The endovascular treatment of carotid aneurysms using covered stents seems to reduce the incidence of peroperative complications, but it may be complicated by asymptomatic thrombosis of the stent.7 Such an approach could not be tried on our patient because of the giant size of aneurysm and the rupture. A perioperative medical treatment associating corticoids and immunosuppressive drugs is always
Vol.
-,
No.
-, -
2015
Case Report 3
Fig. 3. Treatment of the ruptured aneurysm by sternotomy and a cervical approach with a PTFE bypass graft. (A) Appearance after evacuation of aneurysmal thrombus and section of the arterial defect. (B) Control of brachiocephalic artery. (C) Prosthetic graft bypass repair of carotid artery. (D) patient at 2nd postoperative day.
necessary to control the activity of the disease and it seems to reduce some postoperative complications.10
CONCLUSION The arterial manifestations of the BD are serious but seldom inaugural. In our patient, the rupture of a carotid aneurysm revealed the disease. Surgical aneurysmectomy does not always give good long-term results, and carotid ligation was proposed
by several authors as a simple and definitive alternative method, with less late complications. Endovascular exclusion seems to be a promising option, but studies are necessary to evaluate its long-term advantages.
REFERENCES 1. Davidovic L, Dusan K, Maksimovic Z, et al. Carotid artery aneurysms. Vascular 2004;12:166e70.
4 Case Report
2. Ghilardi G, Masseto N, Cattalini C, et al. Brain involvement in extracranial internal carotid artery aneurysms. Vasa 2001;30:37e41. 3. Sayed A, Elwan H, Fouad F, et al. Behcet extracranial carotid aneurysms: is there still a role for ligation? Eur J Vasc Endovasc Surg 2010;39:17e22. 4. Tuzun H, Besirli K, Sayin A, et al. Management of aneurysms in Behcet syndrome: an analysis of 24 patients. Surgery 1997;121:150e6. 5. Ozyazicioglu A, Kocak H, Vural U. Carotid artery pseudoaneurysm in Behcet disease. Eur J Cardiothorac Surg 2001;19:938e9. 6. Kwon Koo B, Shim WH, Yoon YS, et al. Endovascular therapy combined with immunosuppressive treatment for
Annals of Vascular Surgery
7.
8.
9. 10.
pseudoaneurysms in patients with Behcet disease. J Endovasc Ther 2003;10:75e80. Berard X, Corpataux JM, Taoufiq H, et al. Don’t trust a vein graft to treat carotid aneurysm in patients with Behcet disease. Vasc Surg 2010;52:471e4. Kuzu MA, Ozaslan C, Koksoy C, et al. Vascular involvement in Behcet disease: 8-year audit. World J Surg 1994;18: 948e53. Bradbury AW, Milne AA, Murie JA. Surgical aspects of Behcet disease. Br J Surg 1994;81:1712e21. Yekeler E, Tunaci A, Tunaci M, et al. Successful medical treatment of abdominal aortic aneurysms in a patient with Behcet disease: imaging findings. Australas Radiol 2005;49: 182e4.
