A New Therapeutic Approach to Congenital Pelvic Arteriovenous Malformations

A New Therapeutic Approach to Congenital Pelvic Arteriovenous Malformations

A New Therapeutic Approach to Congenital Pelvic Arteriovenous Malformations Rabih Houbballah, Alexandros Mallios, Bertrand Poussier, Patrick Soury, Su...

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A New Therapeutic Approach to Congenital Pelvic Arteriovenous Malformations Rabih Houbballah, Alexandros Mallios, Bertrand Poussier, Patrick Soury, Sumio Fukui, Fre´deric Gigou, and Claude Laurian, Paris, France

Background: Internal iliac arteriovenous malformations (AVM) are difficult to treat. Arterial embolization is chosen in most cases but the angio-architecture of these arteriovenous shunts can provide an explanation for the several reported failures. We report the long-term results of peroperative intravenous embolization. Methods: Between the years 1980 and 2008, seven patients were treated for complex and symptomatic internal AVM. These patients underwent a surgery which involved massive embolization of the venous hypogastric compartment, followed by the ligation of the hypogastric vein at its origin. Results: There were no deaths reported in this group. The mean follow-up was 7 years (range: 10 months-12 years), with no cases of recurrences found. Computed tomographic scans of controls with reconstruction did not show any residual arteriovenous shunts. Conclusion: Intravenous embolization of the internal iliac AVM is a therapeutic strategy which is well adapted to the special angio-architecture of the arteriovenous shunts. Clinical and anatomic results have confirmed the validity of this strategy.

INTRODUCTION Congenital pelvic arteriovenous malformations (AVM) are rare and difficult to treat because of their anatomic location and the multitude of feeding pedicles. Among these malformations, the AVM situated in the anterior territory of the internal iliac artery (IIA) have well-defined anatomic shapes. The evolutive gravity of some of these AVM, leading to regional venous hypertension or cardiac disorders, necessitates aggressive and compulsory surgery.1-3 Previously published data have demonstrated that the use of arterial proximal ligatures as a treatment option was ineffective3-6 and also that selective arterial embolizations are inadequate

because they leave an evolutive residual AVM.2-7 Owing to the difficulty involved in exposure and the resulting severe hemorrhagic complications,4 AVM exeresis as a treatment option is rarely possible. Therefore, we have proposed a mixed approach for the treatment of these AVM which consists of a preoperative embolization of the feeding arterial pedicles combined with a controlled preoperative occlusion of the venous drainage. This procedure which included a nidus occlusion enabled us to obtain a permanent and stable closure of the arteriovenous shunts.

PATIENTS AND METHODS Presented at the 24th Annual Meeting of the French Society for Vascular Surgery, La Rochelle, France, June 2009. Service de Chirurgie Vasculaire, Hoˆpital Saint-Joseph, Paris, France. Correspondence to: Rabih Houbballah, MD, Service de Chirurgie Vasculaire, Hoˆpital Saint-Joseph, rue Raymond Losserand, 75014 Paris, France, E-mail: [email protected] Ann Vasc Surg 2010; 24: 1102-1109 DOI: 10.1016/j.avsg.2010.02.053 Ó Annals of Vascular Surgery Inc.

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A retrospective study of the data obtained from a prospectively completed database was carried out. It included all the patients with symptomatic pelvic AVM who had benefited from a combined arteriovenous treatment at the Saint Joseph Hospital in Paris. Patients underwent a thorough clinical examination which focused on the dominant visceral

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complications (gynecologic, urinary, or colorectal check-up). A cardiac check-up included a transthoracic echography as well as cardiac output measurement. Complementary explorations included either an arterial and venous duplex scan (DS) of the pelvic vessels, an abdominopelvic computed tomographic (CT) scan with reconstruction, or an aortography. A DS of the pelvic vessels identified the main arterial pedicles of the AVM (homolateral and contralateral IIA, inferior mesenteric artery, deep femoral artery) as well as dominant drainage paths (homolateral and/or contralateral internal iliac vein [IIV], deep femoral vein). CT scan along with axial acquisitions and reconstructions in arterial and venous phases allowed for a precise study of the angio-architecture of AVM. A selective and arterial embolization of the anterior IIA trunk was performed without previous ligature. Embolization of the remaining pedicles was not carried out. Surgical Technique A wide exposure of the iliofemoral axis was achieved by performing a median sub-umbilical laparotomy that runs obliquely down toward the iliofemoral homolateral junction. Surgical procedures included ureteral mobilization, IIA dissection with posterior trunk sectioning, and a re-sectioning of the anterior trunk branches so that they are as distally separated as possible (some visceral resections were sometimes found to be necessary, cf. infra). The anterior region of the IIV was exposed and the posterior branches ligatured. Further, the dissection was extended as distally as possible to ligature the inferior venous branches that flow into the deep femoral veins. IIV embolization was carried out after the clamping of the sub-renal aorta and the iliac arteries. The iliac vein was clamped at its extremity to prevent any migration of the embolizing product. Embolization was performed by direct puncture with Ethibloc (Ethicon, Johnson & Johnson, Du¨sseldorf, Germany) and six times with Horsley’s bone wax. The first step of the procedure included liquefying the wax in a bain-marie at 65 C before filling it into sterilized syringes. The product was then injected into the IIV till it was completely filled. After the embolization, the IIV was sutured and the arterial flow restored. A preoperative ultrasound exploration was carried out to detect the presence of any residual shunts. Patients underwent a clinical as well as a radiological follow-up (arterial and venous DS along

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with a CT scan or arteriography as well as postoperative echocardiography at 1, 3, 6 months, and then annually).

RESULTS Between the years 1980 and 2008, seven patients with complex congenital pelvic AVM were treated at our department. Of these, three were women and four were men, mean age was 49 years (range: 37-62), with all of them presenting with symptomatic pelvic AVM (Table I). Isolated or associated symptoms that needed to be treated included cardiac insufficiency with high cardiac output in three patients, hemorrhagic complications (urinary in one patient and digestive in two patients), and dysuria in two patients. Pelvic and supra-pubic pains were reported to be present in all the patients. At our department, mean time difference between the diagnosis of AVM and its treatment was 6.5 years (range: 3 months-40 years). Before being treated at our department, one patient had already benefited from three arterial embolization sessions conducted 7 years ago with clinical and radiological recurrence. Two patients reported having undergone IIA surgical ligature (one bilateral and one unilateral) along with a short-term recurrence. In terms of hemorrhagic symptoms being explored and treated, two patients underwent surgery including an exploratory laparotomy in one patient and a colostomy with rectal packing after hemorrhoid surgery in the other patient. In five patients, the therapeutic sequence started with an initial embolization of the arterial pedicles (two patients did not undergo embolization because of a technical failure and an arterial ligature, respectively). On average, each patient underwent two sessions of therapy. Venous embolization was performed for every patient. A complementary visceral exeresis was carried out in two patients (one hysterectomy with annexectomy to make the approach easier and one AVM resection with partial cystectomy). Postoperative mortality was reported to be nil (Table II). In the first patient to be treated, a severe complication (pulmonary embolism) and two surgical redos (one early and one late) were reported. In fact, this patient did not have any IIV exclusion at the end of the embolization. Immediately after the procedure, he suffered from a moderate pulmonary embolism with migration of the embolization product. He was medically treated without any side-effects. Then, 6 months later, he suffered from

No

No

Normal M 46 7

CF, cardiac flow; Ant Tk, anterior trunk; IIA, internal iliac artery; IMA, inferior mesenteric artery; MSA, median sacral artery.

Two sessions

Homolateral IIA ligature No 46 6

F

Pelvic pain inferior right limb edema Pelvic pain dysuria

Normal

Homolateral Ant Tk IIA Ant Tk IIA bilateral, FCA AntTk IIA Normal Hematuria 37 5

M

Normal 49 4

M

Failure

No No

No No

No 8.5 L/min 50 3

M

62 2

F

Rectorragia after hemorrhoids surgery Cardiac insufficiency Pelvic pain Cardiac insufficiency Pelvic pain dysuria

13 L/min

Ant Tk IIA IMA, bi-ilio femoral IMA MSA Bi-ilio-femoral Ant Tk IIA Bilateral, IMA, MSA Ant Tk IIA 10 L/min 54 1

F

Cardiac insufficiency

AVM arterial pedicles Age (years)

Gender

Symptoms

Cardiac flow

One session Ant Tk IIA One session Ant Tk IIA Two sessions

No

Left colostomy Rectal packing Bilateral IIA ligature

No No No embolization

Annals of Vascular Surgery

Patient

Table I. Preoperative characteristics of the patients

Arterial embolization

Internal iliac ligature artery

Previous surgery

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pelvic heaviness recurrence with radiologically detected residual arteriovenous fistulas and IIV repermeabilization. Redo surgery comprised a new venous embolization using Ethibloc with IIV ligature. At 8 years, this patient suffered again from pelvic heaviness. Arteriography and CT scans did not show any residual fistulas; therefore, the embolized mass was thought to be responsible for this heaviness syndrome. A new (another) surgery with exeresis of the embolized mass brought relief to this patient. At the 12year follow-up, he was asymptomatic with no reported radiological recurrence. Since then, all the other patients underwent embolization with Horsley wax along with ligature of the embolized IIV. Postoperative course was generally simple with blood transfusions of less than three globular units in average. Two patients presented with regressive postoperative crural pains (allegedly because of embolization material diffusion into the IIV posterior branches). None of the patients presented with symptoms associated with the IIV ligature. Mean follow-up was 83 months (range: 10-114 months). Cardiac insufficiency was rapidly regressive with systematic normalization of the cardiac output. On clinical analysis, the AVM was reported as cured. Radiologically (CT scan or arteriography), we did not detect any residual shunts but an IIV thrombosis without any opacification at the early arterial phase (Fig. 4).

DISCUSSION In this part, we will discuss separately the main particularities of the AVM situated in the anterior territory of the IIA (clinical aspects and explorations), the current treatment options, and the angio-architecture of AVM which provided us with the possibility to use our innovative technique. Further, we will discuss the characteristics and results of the technique used in this study. Clinical Aspects The AVM of the anterior internal iliac territory can often be asymptomatic (continuous iliac murmur). Therefore, they cannot be related to any therapy (they do not have to be treated).1,5,8 As for every AVM, the symptoms can be either local or general.6,8 Locally, they result from venous hypertension along with supra-pubic pain risks, pelvic heaviness, compression of the urinary system with obstructive urinary symptoms (dysuria), possible extension to proximate organs with rectorragia,

No No No NR Vein exclusion embolization: 7

6

5

CO, cardiac output; Ant Tk, anterior trunk; IIA, internal iliac artery; IMA, inferior mesenteric artery; MSA, medial sacral artery; CFA, common femoral artery; NR, not reported.

No

No No No Vein exclusion embolization:

NR

96

No

No No No embolization:

NR

80

No

No No 4

Venous wax Venous wax Venous wax Venous wax

embolization:

Partial cystectomy Vein exclusion Vein exclusion

NR

72

Compression by the 144 embolized mass: exeresis No 132 No 5 L/min 3

Venous embolization: wax Venous embolization: Ethibloc 2

No vein exclusion

No

No

Pulmonary embolism: redo surgery for vein exclusion Cruralgia

10 No No

48 No

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No Homolateral regressive crural paralysis No

Hysterectomy 4.5 L/min Annexectomy Left vein exclusion Vein exclusion 4 L/min Venous embolization: wax 1

Surgical exercise Patient Preoperative embolization

Table II. Therapeutic attitude and consequences

Postoperative CO Mortality Complications

Residual AVM Redo surgery

Follow-up/month

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Fig. 1. Aortic CT-scan with 3D reconstruction. Pelvic AVM with right internal iliac vein aneurism (white arrow) with multiple arterial feeding branches including inferior mesenteric artery (black arrow), right median sacral artery and bilateral internal iliac arteries (previously surgically ligated).

hematuria, or meno-metrorragia, which might lead to emergency treatment. It has been commonly acknowledged that an arteriovenous shunt sometimes generates high output cardiac insufficiency. Explorations Arteriographic explorations, although still commonly used, must be replaced with DS and CT scan with reconstructions. Echography must identify the pelvic AVM and the main arterial pedicles. CT scan with reconstructions enables to gather all the information related to the shunt angioarchitecture with axial sections and reconstructions at arterial and venous phases that identify the nidus area around the IIV proximal ectasy (Figs. 1, 2). Therapeutic Alternatives Traditionally, two different treatments have been previously described as therapeutic options.6 AVM surgical exeresis, which is both difficult and dangerous, is one possible therapeutic option.1-4 Surgical difficulties are caused mainly by the flow into the fistula, the high number of feeding vessels, the potential bladder, rectum, uterus or, vesical extensions and the location in the latero-

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Fig. 2. Preoperative aortic CT-scan at the aortic time. Opacification of the Right Internal iliac Vein (black arrow) with multiple feeding arterial pedicles.

rectal cellular space that is more difficult to reach surgically. This deep topography of AVM has often favored arterial occlusion procedures. The artery proximal ligature has in some cases enabled to temporarily reduce the symptoms; however, it needs to be banned because it ineluctably leads to recruit feeding pedicles in other territories and annihilates any other possible arterial embolizations.1-3,5,7 Previously, selective embolization of the IIV anterior branches was a widely used technique to treat a hemorrhagic complication or reduce symptoms.2,9-11 Previously published experiences support the incomplete results of this procedure. Jacobowitz et al.,2 in a series which included 35 patients with a 84-month mean follow-up, reported clinical improvement in 83% of the patients, a lack of improvement in 6%, and aggravation in 9%. This type of treatment requires numerous sessions to be carried out over several months (on an average, three sessions within 24 months). A steady residual radiological AVM with more and more complex angio-architecture was obtained during this treatment. In the published data, Calligaro et al. have confirmed the presence of residual AVM after 80% of arterial embolizations4 (Table III). Therefore, the seriousness of the observed complication, the ineffectiveness of the current

Annals of Vascular Surgery

Fig. 3. Macroscopic pathology view of an Internal Iliac Vein aneurysm involved in a complex pelvic AVM. Note the multiple feeding arterial branches ostias into the vein wall (arrows).

procedures along with the growing complexity of the recurrence treatment, prompted us to seek a therapeutic and curative alternative. But before proposing an AVM curative treatment, we studied the angio-architecture of the AVM. Anatomical Aspects or AngioArchitecture Pelvic AVM appear under different anatomical forms, such as AVM that are extensions of proximal AVM of lower limb, intra-visceral AVM, especially uterine ones and extra-visceral AVM located at the base of the large ligament or in the cellular space of the recto-genito-pubic blades. The AVM of the internal iliac anterior territory are known to belong to the last category. Their angio-architecture is similar to that of arteriovenous fistulas of the intracranial lateral sinus, where the nidus is located in the sinus wall.12,13 At the pelvic level, the nidus is located in the proximal wall of the IIV, where the different feeding pedicles converge (Figs. 1, 3). The arguments in favor of this hypothesis include the initial unilateral nature of the shunts, the proximal ectasy of the iliac vein, the failure of selective arterial embolizations leaving the nidus intact, and the multitude of the pedicles that grow after the arterial ligature or distal embolization (IIA, inferior mesenteric artery, lumbar arteries, medium sacral artery, iliac and

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Treatment of pelvic arteriovenous malformations 1107

Table III. Comparisons with series in the literature Authors (ref)

Technique

Current study

Preoperative percutaneous arterial embolization with preoperative venous embolization Arterial embolization alone (75%) Embolization + surgery (15%) Surgery alone Arterial embolization alone Arterial embolization then surgery Arterial embolization Arterial embolization

Jacobowitz et al2

Calligaro et al4

Castaneda et al9 Game et al7

Number of patients

Mean follow-up (months)

Deaths

Clinical improvement

7

83

0%

100%

35

84

21 10

20 35

2 2

2.8%

Radiological recurrence

0%

83%

NR

5% 0%

47% 40%

67% 80%

50

0%

83%

34%

24 12

0%

100% 100%

NR NR

NR, not reported.

circumflex branches of the deep and common femoral arteries).2,3,7 However, the exeresis of the nidus is most of the time impossible because of this deep anatomical location and associated hemorrhagic risk. Therefore, the embolization of the venous compartment, similar to that proposed in the arteriovenous fistula treatment of the lateral sinus,14 seems to be an interesting strategy. Embolization of the AVM Venous Compartment The percutaneous embolization of the IIV has been described previously;15 however, we deemed it as unsuitable for these high flow AVM because of an important secondary re-impermeabilization risk. In the only case described by Mitsuzaki et al.,15 the preoperative pulmonary embolization risk was prevented by the use of an occlusive balloon placed in the primary iliac vein. This technique represents a risk to secondary embolization at the ablation of the occlusive balloon as was the case in our first patient who did not undergo an IIV ligature. Therefore, our technique was improved by adding the IIV ligature. The preoperative ligature of the venous compartment remains a possible but difficult strategy because of exposure problems and preoperative hemorrhagic risk. The IIV exposure and the control of the drainage paths remain the inevitable pre-requisites to ensure the success of this strategy.

Embolization Material Different materials can be used such as Ethibloc, Horsley’s wax, Ethanol, and Onyx (ev3, Irvine, CA). The latter two materials were not used in this study owing either to the risks involved with their usage (Ethanol) or their cost (Onyx). Ethibloc is a radio-opaque occlusive emulsion. It is a substance obtained by precipitation of the polypeptide after progressive dilution of the ethanol in the blood. Ethibloc becomes solid within 10-15 minutes, thereby obstructing the injected site through a mechanical process.16 It does not easily resorb and can represent a mid-term compressive and impeding mass. Horsley’s bone wax is a hemostatic mixture composed of beeswax (72.5%), paraffin (15%), and isopropyl palmitate (12.5%). Classically, it has been used for the mechanical compression of bone capillaries. The innocuousness of Horsley’s bone wax has been demonstrated especially in cardiac surgery; it is used to perform sternum hemostasis.17 It liquefies between 62 C and 64 C. Its density at 15 C is 0.9580.970 g/cm3. After it becomes a solid, the bone wax causes (generates) a very important inflammatory reaction with constitution of a tissue fibrosis with macrophage and lymphocyte infiltration.18,19 In addition to being low in density and low cost (V1.92 for 2.5 g), this wax has the advantage of hardening in a very short time, being extremely leakproof, it could be well tolerated in the long-term. It can also be extremely well detected under scanning.

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a greater number of patients chosen from different centers with a long-term follow-up (10 years). At present, no other therapeutic alternative enables to cure these complex pelvic AVM. The strategy we propose should be systematically proposed to any patient presenting with a symptomatic and complex pelvic AVM. A percutaneous approach of venous embolization with endoclamping is most likely to become the future of this technique. Its feasibility and efficacy should be explored at short and mid-term intervals.

REFERENCES

Fig. 4. Aortic CT-scan (arterial time) at postoperative day 7. No opacification of the Internal Iliac Vein (black arrow). The internal Iliac Vein is totally occluded by the Horsley’s bone wax.

Analysis of Results The detected complications were related to technical problems. Pulmonary embolism was caused by incomplete IIV exclusion. It is rather difficult to avoid crural pain because it results from the embolization of the superior gluteal vein, which justifies the checking and ligature of the IIV posterior branches before embolization.

CONCLUSION Symptomatic pelvic AVM are complex and difficult to treat. Arterial embolization by microcatheterization remains the first-line of therapy and represents only a palliative treatment with a fairly high clinical and radiological recurrence rate. Venous embolization for (to treat) congenital pelvic AVM is the most suitable therapeutic strategy owing to their specific angio-architecture. Its feasibility and mid-term efficacy have been demonstrated. The reproducibility of this technique will have to be proved by conducting a study that includes

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15. Mitsuzaki K, Yamashita Y, Utsunomiya D, et al. Balloonoccluded retrograde transvenous embolization of a pelvic arteriovenous malformation. Cardiovasc Intervent Radiol 1999;22:518-520. 16. Baud AV, Breton P, Guibaud L, Freidel M. Treatment of lowpressure vascular malformations by injection of Ethibloc. Study of 19 cases and analysis of complications. Rev Stomatol Chir Maxillofac 2000;101:181-188.

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17. Brightmore TG, Hayes P, Humble J, Morgan AD. Haemostasis and healing following median sternotomy. Langenbecks Arch Chir 1975;(Suppl):39-41. 18. Geary JR, Frantz VK. A new absorbable hemostatic bone wax; preliminary report. J Neurosurg 1950;7:383-384. 19. Allison RT. Foreign body reactions and an associated histological artefact due to bone wax. Br J Biomed Sci 1994;51: 14-17.