Papers from the French Vascular Surgery Society 1991 Meeting

Fibromuscular Disease of the External Iliac Artery Andr6 Thevenet, MD, Jean Louis Latil, MD, Bernard Albat, MD, Montpellier, France

Fibromuscular disease is rarely observed in the external lilac artery. During the last 15 years, eight symptomatic cases were encountered in six women and two men whose ages ranged from 29 to 63 years (mean: 47 years). Clinical onset was always recent, either progressive with claudication (three cases) or sudden with abdominal and pelvic pain and acute ischemia due to dissection (five cases). Diagnosis was established by arteriograms showing either a typical appearance of fibromuscular hyperplasia or a segmental dissection or occlusion. Two patients had associated fibromuscular disease of the renal arteries. One of these patients had dysplastic aneurysm of the thyrocervical trunk. Histopathological findings were typical of medial fibromuscular hyperplasia in the seven cases examined. Even though transluminal dilatation is presently simple, adequate, and durable for the management of non-complicated forms, all of our cases were treated surgically either because they were observed before transluminal dilatation was readily available or because of associated dissection. Results of surgery were satisfactory in all cases except one with a mean follow-up of 12.6 years. One patient was reoperated upon 13 years later. (Ann Vasc Surg 1992;6:199-204). KEY WORDS:

lilac artery; fibromuscular disease; arterial dissection; renal artery.

Fibromuscular disease was described by Leadbetter and Burkland [I] in 1938 and has long been considered to be specific to the renal arteries. Several extrarenal sites, including the abdominal, From the Service de Chirurgie Thoracique et Cardiovasculaire, HOpital Aiguelongue, Montpellier, France. Presented at the Annual Meeting of the Soci~tO de Chirurgie Vasculaire de Langue Franqaise, June 21-22, 1991, Marseille, France. Reprint requests: A. Thevenet, MD, Service de Chirurgie Thoracique et Cardiovasculaire, H~pital Aiguelongue, 34059 Montpellier C~dex, France.

cervicocephalic, external iliac, femoral, and popliteal arteries, have been reported since 1964 [2-7]. Fibromuscular disease of the external iliac arteries is rare and usually asymptomatic, and is often discovered accidentally on arteriograms prescribed in work-ups for suspected renovascular hypertension. Between 1973 and 1988, eight symptomatic patients were treated for external iliac artery fibromuscular disease (five bilateral, three unilateral), revealed either by acute ischemia due to dissection or chronic ischemia associated with severe claudication. This series represents 7% of all cases of 199

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TABLE I.mOperated cases of arterial fibromuscular disease (1973-1988) Artery Carotid Renal External iliac Subclavian Celiac Hepatic Superior mesenteric

Number of patients 68 62 8 4 2 2 1

Number of arteries 79 81 13 4 2 2 1

arterial fibromuscular disease operated on during the same interval (Table I).

CASE REPORTS Patient No. 1 Following her fourth delivery, a 48-year-old woman experienced pelvic pain then intermittent claudication of the lower limbs, limiting her walking to 200 meters. Aortograms showed fibromuscular disease of both external iliac arteries. In November 1973, an aortobiiliac prosthetic bypass was performed. After an uneventful postoperative recovery, she has remained asymptomatic for 18 years. Fig. 1. Dissection of right external iliac artery (Patient No. 2). Patient No. 2 A 45-year-old woman was hospitalized on an emergency basis for acute ischemia of the right lower limb in 1974. She had experienced sudden pain in the right flank, iliac fossa and inguinal area, followed by paresthesia in the right lower limb, malaise, and loss of consciousness. A fight femoral pulse was not palpated. Arteriograms were consistent with dissection of the right external iliac artery, as demonstrated by retroperitoneal extravasation of contrast medium (Figs. 1-3). The left external iliac artery was irregular. A prosthetic aortobifemoral bypass associated with right aortoiliac resection was performed. Her postoperative course was uneventful and the patient has remained asymptomatic 17 years later. Patient No. 3 A 63-year-old women, para 2, was hospitalized in December 1974 for bilateral recumbent pain. Five years before she had experienced paresthesia, coldness and fatigability of her lower limbs after walking. Arteriograms showed fibromuscular disease of both external left internal iliac arteries (Figs. 4--6). Circulation was slow distal to that area. Resection-prosthetic right iliac bypass with lumbar sympathectomy was performed. The postoperative course was event free. Three months later, the left iliac artery was resected and a prosthetic bypass inserted, again with lumbar sympathectomy. Clinical and anatomical results were excellent until her death I0 years later

due to medullary compression secondary to Pott's disease. Patient No. 4 A 51-year-old man presented in September 1975 with sudden transient numbness of the left lower limb followed by severe claudication. Three days before he had experienced right lumbar pain with transient hematuria. Arteriograms were consistent with aortoiliofemoral dissection. Three weeks later, repeat arteriograms showed that lesions had decreased and involved both external iliac and the right renal arteries. One month later, resectionanastomosis of the right renal artery was performed along with bilateral iliofemoral prosthetic bypass. The patient's postoperative course was uneventful and he has remained asymptomatic 16 years later. Patient No. 5 A 47-year-old woman, para 4, was h o s p i t a l i z e d for abdominal pain and right iliofemoral bruit in December 1975. Arteriograms documented a pseudo-occlusive stenosis of the right external itiac artery. Local dissection was found at operation. After resection, a prosthetic graft bypass was inserted. Recovery was uneventful, and the patient remains asymptomatic 16 years later.

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Fig. 4. Typical arteriographic aspect of fibromuscular disease of external iliac artery (Patient No. 3). Fig. 2. Dissection associated with thrombosis (Patient No. 2).

Patient No. 6

grams showed fibromuscular disease of both external iliac arteries. Two months later, a right external iliac resection-graft bypass was performed. Her postoperative course was uneventful and results were excellent. In

A 54-year-old woman, para 2, complained of recumbent pain and intermittent claudication of her fight lower limb, starting in August 1978. In September 1978, arterio-

Fig. 3. Fibromuscular disease of media associated with dissection (Patient No. 2).

Fig. 5. Gross aspect: alternation of dilatations and fibrous septa (Patient No. 3).

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Fig. 6. Fibromuscular disease of media (Patient No. 3). 1983, hypertension was discovered and repeat artefiograms documented fibromuscular disease of both renal arteries. Again, resection and prosthetic bypass was performed. The patient remained asymptomatic for 13 years. In 1991, right lower limb claudication appeared. Arteriograms showed that the external iliac bypass was occluded. A femorofemoral prosthetic crossover bypass was performed with satisfactory early results. Patient No. 7 A 29-year-old woman, para 2, had left iliac fossa pain and impaired walking in 1988. Digital subtraction arteriograms showed dissection of the left external iliac artery and an aneurysm of the left thyrocervical trunk (Fig. 7). A vein bypass was inserted after resection of the left external iliac artery, and obliterative endoaneurysmorrhaphy of the left thyrocervical trunk was performed. The postoperative course was uneventful. This patient has remained asymptomatic for three years with another pregnancy. Patient No. 8

A 53-year-old man experienced intermittent claudication of his right lower limb, with claudication distance

ANNALS OF V A S C U L A SURGERY R

Fig. 7. Dissection associated with thrombosis of left external lilac artery (Patient No. 7).

limited to 300 meters in July 1988. Arteriograms showed dissection of the right external iliac artery. A fight iliofemoral prosthetic bypass was inserted after excision of the external iliac artery. This patient's postoperative course was uneventful, and he remained asymptomatic until he died two years later of acute pancreatitis.

DISCUSSION F i b r o m u s c u l a r disease of the external iliac artery is a rare disease whose exact incidence is unknown. Of 70 patients with fibromuscular disease of the renal arteries, Wylie and associates [5] found four instances (5.7%) of a s y m p t o m a t i c , external iliac artery fibromuscular disease. External iliac artery fibromuscular disease is often associated with fibromuscular disease o f the renal or another artery. In our series, this was the case in three patients, as there were two instances of renal artery fibromuscular disease and one dysplastic a n e u r y s m of the thyrocervical trunk. In a series of 12 patients, Walter and colleagues [8] o b s e r v e d five cases of

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bilateral renal dysplasia and four cases of unilateral renal dysplasia. The typical radiological findings include successive annular stenotic segments alternating with interstrictural dilatations. An occasional tubular stenosis probably indicates dissection, which can equally resemble occlusion. In all our cases, there was fibromuscular disease of the media characterized by dissociation of the media by fibrous material producing caliber irregularity with the presence of mural microaneurysms. This fibrous proliferation is accompanied by altered muscular fibers which are dispersed, as well as degraded, notched, fragmented, doubled, or atrophied. The intima is normal while the adventitia is inflammatory. At operation, the external iliac arteries appear too long with successive alternating strictures and dilatations. The origin of fibromuscular disease is unknown. The predominance of women sufferers suggests a hormonal influence on smooth muscle fibers. Genetic factors or abnormal traction on the arterial wall have also been suggested. Our series included six women, all of whom had several pregnancies. The mean age of patients was 47 years (range 29 to 63 years). None of these patients had any risk factors for atheroma. The clinical picture can be one of arterial occlusive disease, peripheral embolism, or dissection. The narrowed arterial lumen leads to decreased perfusion pressure and is responsible for symptoms of peripheral chronic occlusive arterial disease. Intermittent claudication is rapidly progressive with abolition of or diminished pulsations in the lower limbs and iliofemoral bruit. Turbulence and blood stasis at the level of interstrictural dilatations suggest platelet aggregation and the formation of microthrombi, possibly at the origin of peripheral microembolism to the toes [9]. This was not observed in our series. In our series, three patients had intermittent claudication and five had dissection. This last complication, which can lead to acute or subacute ischemia, has rarely been reported [6,10]. Onset is characterized by abdominal, pelvic, and femoral pain associated with coldness of the lower limb. The dissection compresses the arterial lumen and can result in occlusion of the external iliac artery. Arteriographically-proven spontaneous restoration of patency of a dissected external iliac artery at four months was observed by Sauer and coworkers [10]. Symptomatic external iliac artery fibromuscular disease warranting treatment has been reported only sporadically in the literature. In 1966, Najafi [11] reported the first case of bilateral external iliac artery fibromuscular disease treated by resectionvenous graft. In 1971, Pollit and associates [12] described a similar case, but the patient refused operation. The chronological list of symptomatic patients

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TABLE II.--External lilac artery fibromuscular disease (review of the literature) First author (reference) Najafi [11] Home [13] Walter [8] Daskalaskis [14] Houston [15] Burri [16] Parnell [17] Sauer [1 O] Present series

Year

Number of patients

1966 1975 1978 1978

1 1 1 1

1979 1983 1988 1990

1 t 1 4 3 8

199t

Treatment Surgery Dilatation + + + + + + + + + +

undergoing surgical correction is presented in Table II. These include surgical revascularization [8,10, 11,13,16, this series], intraoperative [10,14,15] or femoral percutaneous [17] transluminal dilatation. Transluminal dilatation has been shown to be durable in carotid or renal fibromuscular disease in large series. Six of the patients with external iliac artery fibromuscular disease treated by dilatation (five by intraoperative dilatation) are free of symptoms four to seven years later. Surgical access to the external iliac artery can be gained through the transperitoneal, retroperitoneal or femoral route. The reported revascularization procedures included one resection-anastomosis, five combined resection-grafts (one vein, one polytetrafluoroethylene [PTFE], three Dacron), and two aortofemoral Dacron bypasses. In our series of eight operated patients, presently the largest series published, a total of 13 external iliac arteries with fibromuscular disease were restored by two aortobifemoral bypasses, three iliofemoral bypasses, and six external iliac artery resection-graftings. As all symptoms have disappeared on follow-ups ranging from two to 18 years (mean 12.6 years, median 15 years), our results can be considered as satisfactory. There were two late deaths in this series, one due to acute pancreatitis (two years later) and the other due to medullary compression by Port's disease (10 years later). Three patients underwent simultaneous revascularization procedures to the renal artery. One of them also had treatment of a thyrocervical aneurysm. One patient underwent PTFE replacement of both renal arteries for renovascular hypertension due to fibromuscular disease five years after operation on the external iliac artery. The external iliac artery occluded 13 years later with repeat short distance claudication, and it was treated by crossover femorofemoral bypass, resulting in the disappearance of symptoms. Indications for surgical treatment depend upon symptoms. Chronic ischemia associated with clau-

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dication should be treated by dilatation, preferably through the surgical femoral route. This method has been shown to be simple, adequate, and safe. On the other hand, patients with peripheral microembolisms and, above all, dissection and aneurysm should be treated surgically by resection of the external iliac artery followed by reestablishment of continuity by prosthetic bypass.

8. 9. 10. I1. 12.

REFERENCES 1. LEADBETTER WF, BURKLAND CE. Hypertension in unilateral renal disease. J Urol 1938;39:611. 2. PALUBINSKAS AJ, PERLOFF D, NEWTON TH. Fibromuscular hyperplasia in extrarenat arteries. Radiology 1964; 82:451-455. 3. PALUBINSKAS AJ, PERLOFF D, NEWTON TH. Fibromuscular hyperplasia: an arterial dysplasia of increasing clinical importance. A JR 1966;98:907-913. 4. TWlGG HL, PALMISANO PJ. Fibromuscular hyperplasia of the iliac artery. A m J Roentgenol 1965;95:418--423. 5. WYLIE EJ, BINKLEY FM, PALUBINSKAS AJ. Extrarenal fibromuscular hyperplasia. A m J Surg 1966;112:149-154. 6. T H E V E N E T A, MARY H. Dysplasies et dissections des art~res iliaques externes. In: Actualit~s Chirurgicales. Paris: Masson, 1977, pp. 17-19. 7. IWAI T, KONNO S, HIEIJIMA K, et al. Fibromuscular

13. 14. 15.

16. 17.

mmnnu

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dysplasia in the extremities. J Cardiovasc Surg 1985;26.'495501. WALTER JF, STANLEY JC, MEHIGAN JT, et al. External iliac artery fibrodysplasia. A JR 1978;131:125-128. MEHIGAN JT, STONEY RJ. Arterial microemboli and fibromuscular dysplasia of the external iliac arteries. Surgery 1977 ;81:484--486. SAUER L, REILLY LM, GOLDSTONE J, et al. Clinical spectrum of symptomatic external iliac fibromuscular dysplasia. J Vasc Surg 1990;12:488-496. NAJAFI H. Fibromuscular hyperplasia of external iliac artery. Arch Surg 1966;92:394-396. POLLIT J, BOLINO A, KURRAL JC. Symptomatic fibromuscular hyperplasia of external iliac artery. Vasc Surg 1971;5:115-119. HORNE TW. Fibromuscular hyperplasia of the iliac arteries. Aust N Z J Surg 1975;45:415-417. DASKALASKIS MK. Fibromuscular hyperplasia of external iliac arteries. West J Med 1978;128:345-347. HOUSTON C, ROSENTHAL D, LAMIS PA, et al. Fibromuscular dysplasia of the external iliac arteries: surgical treatment by gradual internal dilatation technique. Surgery 1979;85:713-715. BURRI B, FONTOLLIET C, RUEGSEGGER GH, et al. External iliac artery dissection due to fibromuscular dysplasia. Vasa 1983;12:76-78. PARNELL AP, LOOSE AW, CHAMBERLAIN J. Fibromuscular dysplasia of the external iliac artery treatment by percutaneous transluminal angioplasty. Br J Radiol 1988;61: t080-1082.

Fibromuscular disease of the external iliac artery.

Fibromuscular disease is rarely observed in the external iliac artery. During the last 15 years, eight symptomatic cases were encountered in six women...
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