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Surgical Repair of a Giant Isolated Inflammatory Aneurysm of the Left Common Iliac Artery
Surgical Repair of a Giant Isolated Inflammatory Aneurysm of the Left Common Iliac Artery Marcus Fokou,1 Abel Teyang,1 Emmanuel Fongang,2 Justin Kamga,1 Fidele Binam,3 and Wilhem Sandmann,4 Yaounde, Cameroon and D€ usseldorf, Germany
Isolated aneurysms of the iliac artery (IA) are very rare. Inflammatory aneurysms are also rare conditions, making the association rarer. Only four cases of isolated inflammatory IA aneurysms have been reported in the last decade. In this article, we report a case of a 62-year-old patient with a 13-cm large isolated inflammatory aneurysm of the left common IA associated with a 2.5-cm right common IA involvement. The patient presented with a left lower quadrant pain, a pulsatile mass with bruit, as well as a left hydronephrosis secondary to the ipsilateral ureter entrapment, and a motor and sensory deficit of the left lower extremity. An aortoiliofemoral prosthetic repair associated to a left to right ureteral transposition was undertaken with an approach including a laparotomy and bilateral femoral artery exposure. The postoperative period was uneventful. The case mentioned in this study is probably the largest IA aneurysm ever described.
Isolated aneurysms of the iliac arteries (IAs), be it the common iliac artery (CIA), internal iliac artery (IIA), or external iliac artery, are rare but well-documented diseases.1,2 Most of the aneurysms of the iliac system are degenerative and are seen in association with an abdominal aortic aneurysm. It is thought to occur in about 1-7% of the incidence of abdominal aortic aneurysms.1,2 At the same time, inflammatory aneurysms represent only 5-10% of all aneurysms encountered. Therefore, the association is even rarer and only four cases of isolated inflammatory IA aneurysms have been published during the last decade.3-5 In this study, we present a challenging and rare case of a patient with a 13-cm large isolated inflammatory 1
Department of Surgery, Yaounde General Hospital, Yaounde, Cameroon. 2 Department of Radiology, Yaounde General Hospital, Yaounde, Cameroon. 3 Department of Anesthesiology, Yaounde General Hospital, Yaounde, Cameroon. 4 Department of Vascular Surgery and Kidney Transplantation, University D€ usseldorf, D€ usseldorf, Germany.
Correspondence to: Marcus Fokou, MD, Department of Vascular and Transplantation Surgeon H^opital General de Yaounde, BP 5408 Yaounde, Cameroun, E-mail: [email protected] Ann Vasc Surg 2011; 25: 700.e1-700.e4 DOI: 10.1016/j.avsg.2010.09.030 Ó Annals of Vascular Surgery Inc. Published online: April 21, 2011
aneurysm of the left CIA with a 2.5-cm right CIA involvement. The patient was treated surgically. To the best of our knowledge, this is the largest IA aneurysm ever described.
CASE REPORT The patient, a 62-year-old farmer, was examined in the Surgical Department of the Yaounde General Hospital in Cameroon. He presented with a 3-month history of a gradually increasing painful and swelling lower left abdominal quadrant. He was a nonsmoker and a moderate alcohol consumer. No other past medical disease or vascular risk factor was reported. He developed a left lower extremity weakness and a slight edema. He was referred to our vascular department because an abdominal ultrasound revealed the vascular nature of the mass. On examination, the mass which measured 15 cm was firm, slightly painful, pulsatile, and a bruit was easily audible on auscultation (Fig. 1). The left leg examination revealed a reduced muscular strength (3/5) and a hypoesthesia of the anterior side of the thigh. The pulsation was palpable on rectal examination. The computed tomography angiography work-up (Figs. 2 and 3) demonstrated a left CIA aneurysm. Its anteroposterior diameter was 13.3 cm, and the cephalocaudal length 21 cm. A right 2.2-cm CIA aneurysm was also present. There was no evidence of systemic atherosclerosis. The left hypogastric artery was not patent but the right was. A left hydroureteronephrosis secondary to ureteral entrapment was also 700.e1
700.e2 Case reports
Annals of Vascular Surgery
Fig. 1. Clinical aspect of the giant left iliac artery (IA) aneurysm.
Fig. 3. Left common IA aneurysm demonstrating the patency of the femoral arteries and the hydronephrosis (arrow).
Fig. 2. Left 13.3-cm IA aneurysm with a huge thrombus (long arrow) and right 2.2-cm common IA aneurysm (short arrow). demonstrated. A cardiac examination was conducted but there was no evidence of heart disease. With regard to laboratory investigations, an inflammatory syndrome was present (erythrocyte sedimentation rate: 75 mm/H), the renal function test was normal (creatinine: 8 mg/ dL), and the urine culture sterile. The patient was human immunodeficiency virus (HIV) negative. The other preoperative work-ups showed no abnormality. Under general and epidural anesthesia, exposure of the aneurysm was realized proximally through a midline laparotomy and distally through a bilateral femoral exposure. The sigmoid, the mesentery, and the urinary bladder were partly involved in the fibrosis. They were also deviated and compressed. The left ureteral entrapment was severe and extended to its middle segment. We decided
to perform a graft insertion technique using a bifurcation 16/8/8-mm Dacron graft. Before the 65 minutes of the clamping time, 5,000 IU heparin was administered intravenously. After the opening of the aneurysmal sac, its wall was found to be hard and fibrous. An end-to-end anastomosis with the infrarenal aorta was performed. Distally, a left common femoral anastomosis was performed and the right branch of the graft was inserted through the CIA, therefore preserving the right IIA. Ureterolysis was impossible because of the extent of the fibrous tissue. Therefore, a left to right ureteral transposition was realized. The sigmoid and other adhesions were left in place. The postoperative period was uneventful. No ischemia was noticed in the lower limbs. The renal function test was normal. The patency was assessed clinically and with ultrasound daily till discharge from the hospital (2 weeks), then monthly for one trimester, and every 6 months since then. A biopsy of the aneurysmal sac was taken. Histologically, there was no evidence of atherosclerosis but a characteristic perivascular inflammation was present. As of date, 14 months after the operation, the state of circulation in the lower limbs is good, the renal function is normal, the left leg weakness and swelling have improved, and there is no sexual dysfunction.
DISCUSSION Epidemiology Isolated inflammatory IA aneurysms are extremely rare. There is much debate about the etiology of
Vol. 25, No. 5, July 2011
inflammatory aneurysms because the inciting event initiating the inflammatory response is unknown.3-5 Similar to our patient, another isolated IA aneurysm, although not inflammatory, has been reported in Africa, a region where vascular diseases are mistakenly considered to be inexistent.6 Most patients with inflammatory aneurysms described in the aorta or as isolated IA aneurysms are men in their 70s with other vascular risk factors, contrary to our patient.1,2 Uncommon aneurysms in size, topography, and evolution have been described in relation to HIV.7 Fortunately, our patient was HIV negative. The CIA is mostly involved and bilateral aneurysms are encountered in about 27% of patients with isolated IA aneurysms, as in our patient.1,2 Manifestations The natural history of isolated inflammatory IA aneurysms is not well known because of the paucity of experience. Most IA aneurysms, whether involving the CIA, IIA, or external iliac artery, are asymptomatic and are most often detected on imaging studies conducted for unrelated reasons. However, it may be occasionally suspected in case of local signs and compressive complications, as seen in the patient presented in this study.1-5 In the few publications about IA inflammatory aneurysms, like in our patient, a lower quadrant pain was present. The latter can mimic appendicitis when involving the right side, as reported by Mieog et al.3 Weakness and the hypoesthesia of the left leg are unusual. Huge iliac aneurysms may present some unusual presentations like these. It is presumably because of lumbar plexus nerves compression by the aneurysmal mass and their involvement in the perivascular fibrosis. In inflammatory aneurysms, a characteristic thickened wall is generally seen on computed tomographic scan. But, in this study, it was not clearly demonstrated on computed tomography. However, the preoperative findings as well as the macroscopic and histologic aspects definitively confirm the diagnosis. Treatment Treatment recommendations have been mostly based on anecdotal impression rather than on a large collective experience. Intervention is recommended for all symptomatic IA aneurysms. A diameter of >3 cm is the threshold in asymptomatic atherosclerotic IA aneurysms.1,2 Because of its rarity, the natural history of inflammatory
Case reports 700.e3
aneurysm is unknown; therefore, a threshold is difficult to define. But because of the high risk of rupture and adjacent structures entrapment, surgical management at a smaller diameter is perhaps the best choice. Several types of reconstructions have been proposed on the basis of the size, location, and etiology of the aneurysm, but the increasing availability of stent-grafts has greatly extended the treatment options.2 The most common is the resection of the aneurysm with reconstruction of the artery using a prosthetic material. Other options include a bifurcated prosthesis, aneurysmorrhaphy, and reconstruction using a venous or prosthetic patch.1,2 A ligation of the IIA or a ‘‘banding’’ has also been performed by many authors.1,2 The management of the entrapped ureter is controversial in aortic artery inflammatory aneurysms. Although most authorities advise a nonaggressive attitude assuming that the inflammation will resolve after the aneurysm is cured, others recommend an ureterolysis in cases when the entrapment is very much extended. But in IA inflammatory aneurysms, no recommendation is clearly available because of the extreme rarity of the entity and the smaller size of the previously published cases.3,4 In this case, the extent of the fibrosis urged us to attempt to liberate the ureter. This was technically difficult, so we finally performed a ureteral transposition. Surgery is the traditional definitive treatment, although the endovascular therapy with stentgrafts is increasingly used.1,2 Recently Patel et al. reported a 12-year experience with endovascular and open repair in the management of isolated IA aneurysms. The proportion of open repair was 24 of 56 patients.2 Embolization as well as coiling could be alternatives in treating IIA aneurysms. As mentioned previously, no specific therapeutic option is proposed for inflammatory IA because of its rarity. In our patient, we performed a reconstruction using a bifurcated Dacron prosthesis. Because there was a high operative risk in the case of this patient, a stent-graft was also discussed; however, an endovascular approach as initial treatment modality was excluded for the following reasons. First, the high risk of endoleak related to the huge size of the aneurysm. Because the compression of the neighboring organs was very pronounced, an endoleak could have somewhat maintained the compression. Second, the right CIA was involved in the aneurysmal process and only the right hypogastric artery was patent. It would have been very difficult to insert a bifurcated stent while preserving it.
700.e4 Case reports
The surgical exposure was a midline laparotomy and bilateral groin incisions. Other possibilities such as a retroperitoneal flank incision, laparotomy without femoral exposures but internal and external iliac vessels preparation, or a laparotomy with an endovascular occlusion after the incision of the aneurysmal sac were discussed. But considering that the anatomy of the region was distorted by the large size of the lesion, its closed contact to the pelvic wall, and the significant aneurysm of the right CIA, we thought that the initial control of the aorta and the inferior side of the aneurysm and distal vessels were imperative to avoid excessive bleeding. Only a triple exposure could permit us to prevent this complication and insert the graft accordingly.
CONCLUSION Despite its clinical magnitude, isolated inflammatory IA aneurysms are uncommon and the published experience with this condition is limited. A surgical approach including a bilateral femoral
Annals of Vascular Surgery
exposure and a ureteral transposition can be helpful to control a huge aneurysm. REFERENCES 1. Ferreira J, Canedo A, Brandao D, et al. Isolated iliac artery aneurysms: six- year experience. Interact Cardiovasc Thorac Surg 2010;10:245-248. 2. Patel NV, Long GW, Cheema ZF, Rimar K, Brown OW, Shanley CJ. Open vs. endovascular repair of isolated iliac artery aneurysms: a 12-year experience. J Vasc Surg 2009;49:1147-1153. 3. Mieog JS, Stoot JH, Bosch JJ, Koning OH, Hamming JF. Inflammatory aneurysm of the common iliac artery mimicking appendicitis. Vascular 2008;16:116-119. 4. Magliocca JF, Faerber GJ, Upchurch GR Jr. Solitary common iliac artery inflammatory aneurysm in a healthy woman: case report and review of the literature. Ann Vasc Surg 2005;19: 890-895. 5. Shindo S, Kubota K, Kojima A, et al. Inflammatory solitary iliac artery aneurysms: a report of two cases. Cardiovasc Surg 2001;9:615-619. 6. Hodasi WM. Isolated internal iliac artery aneurysm and urinary retention. West Afr J Med 1995;14:184-185. 7. Nair R, Robbs JV, Naidoo NG, Woolgar J. Clinical profile of HIV-related aneurysms. Eur J Vasc Endovasc Surg 2000;20: 235-240.
Isolated aneurysms of the iliac artery (IA) are very rare. Inflammatory aneurysms are also rare conditions, making the association rarer. Only four cases of isolated inflammatory IA aneurysms have been reported in the last decade. In this article, we report a case of a 62-year-old patient with a 13-cm large isolated inflammatory aneurysm of the left common IA associated with a 2.5-cm right common IA involvement. The patient presented with a left lower quadrant pain, a pulsatile mass with bruit, as well as a left hydronephrosis secondary to the ipsilateral ureter entrapment, and a motor and sensory deficit of the left lower extremity. An aortoiliofemoral prosthetic repair associated to a left to right ureteral transposition was undertaken with an approach including a laparotomy and bilateral femoral artery exposure. The postoperative period was uneventful. The case mentioned in this study is probably the largest IA aneurysm ever described.
Isolated aneurysms of the iliac arteries (IAs), be it the common iliac artery (CIA), internal iliac artery (IIA), or external iliac artery, are rare but well-documented diseases.1,2 Most of the aneurysms of the iliac system are degenerative and are seen in association with an abdominal aortic aneurysm. It is thought to occur in about 1-7% of the incidence of abdominal aortic aneurysms.1,2 At the same time, inflammatory aneurysms represent only 5-10% of all aneurysms encountered. Therefore, the association is even rarer and only four cases of isolated inflammatory IA aneurysms have been published during the last decade.3-5 In this study, we present a challenging and rare case of a patient with a 13-cm large isolated inflammatory 1
Department of Surgery, Yaounde General Hospital, Yaounde, Cameroon. 2 Department of Radiology, Yaounde General Hospital, Yaounde, Cameroon. 3 Department of Anesthesiology, Yaounde General Hospital, Yaounde, Cameroon. 4 Department of Vascular Surgery and Kidney Transplantation, University D€ usseldorf, D€ usseldorf, Germany.
Correspondence to: Marcus Fokou, MD, Department of Vascular and Transplantation Surgeon H^opital General de Yaounde, BP 5408 Yaounde, Cameroun, E-mail: [email protected] Ann Vasc Surg 2011; 25: 700.e1-700.e4 DOI: 10.1016/j.avsg.2010.09.030 Ó Annals of Vascular Surgery Inc. Published online: April 21, 2011
aneurysm of the left CIA with a 2.5-cm right CIA involvement. The patient was treated surgically. To the best of our knowledge, this is the largest IA aneurysm ever described.
CASE REPORT The patient, a 62-year-old farmer, was examined in the Surgical Department of the Yaounde General Hospital in Cameroon. He presented with a 3-month history of a gradually increasing painful and swelling lower left abdominal quadrant. He was a nonsmoker and a moderate alcohol consumer. No other past medical disease or vascular risk factor was reported. He developed a left lower extremity weakness and a slight edema. He was referred to our vascular department because an abdominal ultrasound revealed the vascular nature of the mass. On examination, the mass which measured 15 cm was firm, slightly painful, pulsatile, and a bruit was easily audible on auscultation (Fig. 1). The left leg examination revealed a reduced muscular strength (3/5) and a hypoesthesia of the anterior side of the thigh. The pulsation was palpable on rectal examination. The computed tomography angiography work-up (Figs. 2 and 3) demonstrated a left CIA aneurysm. Its anteroposterior diameter was 13.3 cm, and the cephalocaudal length 21 cm. A right 2.2-cm CIA aneurysm was also present. There was no evidence of systemic atherosclerosis. The left hypogastric artery was not patent but the right was. A left hydroureteronephrosis secondary to ureteral entrapment was also 700.e1
700.e2 Case reports
Annals of Vascular Surgery
Fig. 1. Clinical aspect of the giant left iliac artery (IA) aneurysm.
Fig. 3. Left common IA aneurysm demonstrating the patency of the femoral arteries and the hydronephrosis (arrow).
Fig. 2. Left 13.3-cm IA aneurysm with a huge thrombus (long arrow) and right 2.2-cm common IA aneurysm (short arrow). demonstrated. A cardiac examination was conducted but there was no evidence of heart disease. With regard to laboratory investigations, an inflammatory syndrome was present (erythrocyte sedimentation rate: 75 mm/H), the renal function test was normal (creatinine: 8 mg/ dL), and the urine culture sterile. The patient was human immunodeficiency virus (HIV) negative. The other preoperative work-ups showed no abnormality. Under general and epidural anesthesia, exposure of the aneurysm was realized proximally through a midline laparotomy and distally through a bilateral femoral exposure. The sigmoid, the mesentery, and the urinary bladder were partly involved in the fibrosis. They were also deviated and compressed. The left ureteral entrapment was severe and extended to its middle segment. We decided
to perform a graft insertion technique using a bifurcation 16/8/8-mm Dacron graft. Before the 65 minutes of the clamping time, 5,000 IU heparin was administered intravenously. After the opening of the aneurysmal sac, its wall was found to be hard and fibrous. An end-to-end anastomosis with the infrarenal aorta was performed. Distally, a left common femoral anastomosis was performed and the right branch of the graft was inserted through the CIA, therefore preserving the right IIA. Ureterolysis was impossible because of the extent of the fibrous tissue. Therefore, a left to right ureteral transposition was realized. The sigmoid and other adhesions were left in place. The postoperative period was uneventful. No ischemia was noticed in the lower limbs. The renal function test was normal. The patency was assessed clinically and with ultrasound daily till discharge from the hospital (2 weeks), then monthly for one trimester, and every 6 months since then. A biopsy of the aneurysmal sac was taken. Histologically, there was no evidence of atherosclerosis but a characteristic perivascular inflammation was present. As of date, 14 months after the operation, the state of circulation in the lower limbs is good, the renal function is normal, the left leg weakness and swelling have improved, and there is no sexual dysfunction.
DISCUSSION Epidemiology Isolated inflammatory IA aneurysms are extremely rare. There is much debate about the etiology of
Vol. 25, No. 5, July 2011
inflammatory aneurysms because the inciting event initiating the inflammatory response is unknown.3-5 Similar to our patient, another isolated IA aneurysm, although not inflammatory, has been reported in Africa, a region where vascular diseases are mistakenly considered to be inexistent.6 Most patients with inflammatory aneurysms described in the aorta or as isolated IA aneurysms are men in their 70s with other vascular risk factors, contrary to our patient.1,2 Uncommon aneurysms in size, topography, and evolution have been described in relation to HIV.7 Fortunately, our patient was HIV negative. The CIA is mostly involved and bilateral aneurysms are encountered in about 27% of patients with isolated IA aneurysms, as in our patient.1,2 Manifestations The natural history of isolated inflammatory IA aneurysms is not well known because of the paucity of experience. Most IA aneurysms, whether involving the CIA, IIA, or external iliac artery, are asymptomatic and are most often detected on imaging studies conducted for unrelated reasons. However, it may be occasionally suspected in case of local signs and compressive complications, as seen in the patient presented in this study.1-5 In the few publications about IA inflammatory aneurysms, like in our patient, a lower quadrant pain was present. The latter can mimic appendicitis when involving the right side, as reported by Mieog et al.3 Weakness and the hypoesthesia of the left leg are unusual. Huge iliac aneurysms may present some unusual presentations like these. It is presumably because of lumbar plexus nerves compression by the aneurysmal mass and their involvement in the perivascular fibrosis. In inflammatory aneurysms, a characteristic thickened wall is generally seen on computed tomographic scan. But, in this study, it was not clearly demonstrated on computed tomography. However, the preoperative findings as well as the macroscopic and histologic aspects definitively confirm the diagnosis. Treatment Treatment recommendations have been mostly based on anecdotal impression rather than on a large collective experience. Intervention is recommended for all symptomatic IA aneurysms. A diameter of >3 cm is the threshold in asymptomatic atherosclerotic IA aneurysms.1,2 Because of its rarity, the natural history of inflammatory
Case reports 700.e3
aneurysm is unknown; therefore, a threshold is difficult to define. But because of the high risk of rupture and adjacent structures entrapment, surgical management at a smaller diameter is perhaps the best choice. Several types of reconstructions have been proposed on the basis of the size, location, and etiology of the aneurysm, but the increasing availability of stent-grafts has greatly extended the treatment options.2 The most common is the resection of the aneurysm with reconstruction of the artery using a prosthetic material. Other options include a bifurcated prosthesis, aneurysmorrhaphy, and reconstruction using a venous or prosthetic patch.1,2 A ligation of the IIA or a ‘‘banding’’ has also been performed by many authors.1,2 The management of the entrapped ureter is controversial in aortic artery inflammatory aneurysms. Although most authorities advise a nonaggressive attitude assuming that the inflammation will resolve after the aneurysm is cured, others recommend an ureterolysis in cases when the entrapment is very much extended. But in IA inflammatory aneurysms, no recommendation is clearly available because of the extreme rarity of the entity and the smaller size of the previously published cases.3,4 In this case, the extent of the fibrosis urged us to attempt to liberate the ureter. This was technically difficult, so we finally performed a ureteral transposition. Surgery is the traditional definitive treatment, although the endovascular therapy with stentgrafts is increasingly used.1,2 Recently Patel et al. reported a 12-year experience with endovascular and open repair in the management of isolated IA aneurysms. The proportion of open repair was 24 of 56 patients.2 Embolization as well as coiling could be alternatives in treating IIA aneurysms. As mentioned previously, no specific therapeutic option is proposed for inflammatory IA because of its rarity. In our patient, we performed a reconstruction using a bifurcated Dacron prosthesis. Because there was a high operative risk in the case of this patient, a stent-graft was also discussed; however, an endovascular approach as initial treatment modality was excluded for the following reasons. First, the high risk of endoleak related to the huge size of the aneurysm. Because the compression of the neighboring organs was very pronounced, an endoleak could have somewhat maintained the compression. Second, the right CIA was involved in the aneurysmal process and only the right hypogastric artery was patent. It would have been very difficult to insert a bifurcated stent while preserving it.
700.e4 Case reports
The surgical exposure was a midline laparotomy and bilateral groin incisions. Other possibilities such as a retroperitoneal flank incision, laparotomy without femoral exposures but internal and external iliac vessels preparation, or a laparotomy with an endovascular occlusion after the incision of the aneurysmal sac were discussed. But considering that the anatomy of the region was distorted by the large size of the lesion, its closed contact to the pelvic wall, and the significant aneurysm of the right CIA, we thought that the initial control of the aorta and the inferior side of the aneurysm and distal vessels were imperative to avoid excessive bleeding. Only a triple exposure could permit us to prevent this complication and insert the graft accordingly.
CONCLUSION Despite its clinical magnitude, isolated inflammatory IA aneurysms are uncommon and the published experience with this condition is limited. A surgical approach including a bilateral femoral
Annals of Vascular Surgery
exposure and a ureteral transposition can be helpful to control a huge aneurysm. REFERENCES 1. Ferreira J, Canedo A, Brandao D, et al. Isolated iliac artery aneurysms: six- year experience. Interact Cardiovasc Thorac Surg 2010;10:245-248. 2. Patel NV, Long GW, Cheema ZF, Rimar K, Brown OW, Shanley CJ. Open vs. endovascular repair of isolated iliac artery aneurysms: a 12-year experience. J Vasc Surg 2009;49:1147-1153. 3. Mieog JS, Stoot JH, Bosch JJ, Koning OH, Hamming JF. Inflammatory aneurysm of the common iliac artery mimicking appendicitis. Vascular 2008;16:116-119. 4. Magliocca JF, Faerber GJ, Upchurch GR Jr. Solitary common iliac artery inflammatory aneurysm in a healthy woman: case report and review of the literature. Ann Vasc Surg 2005;19: 890-895. 5. Shindo S, Kubota K, Kojima A, et al. Inflammatory solitary iliac artery aneurysms: a report of two cases. Cardiovasc Surg 2001;9:615-619. 6. Hodasi WM. Isolated internal iliac artery aneurysm and urinary retention. West Afr J Med 1995;14:184-185. 7. Nair R, Robbs JV, Naidoo NG, Woolgar J. Clinical profile of HIV-related aneurysms. Eur J Vasc Endovasc Surg 2000;20: 235-240.