A True Giant Aneurysm of the Anterior Tibial Artery Debora Musio, Maria Cecilia Perfumo, Valerio Gazzola, Bianca Pane, Giovanni Spinella, and Domenico Palombo, Genova, Italy

Aneurysms of the anterior tibial artery are rare. We will describe a case of a woman with an asymptomatic true aneurysm of the anterior tibial artery. The patient presented with a pulsatile mass in the lateral face of the distal portion of the left leg, and both ultrasound examination and computed tomography angiography scan showed a giant aneurysm of the anterior tibial artery. We chose open treatment.

Aneurysms of lower limb arteries are a rare pathology and the most common causes are trauma of the lower limbs, infections (either localized or systemic), and collagen disorders. The anterior tibial artery (ATA) is a very rare localization for aneurysmal dilation.1e8 We will describe a case of a female patient with an asymptomatic aneurysm located on the medial part of the left ATA.

CASE REPORT A 63-year-old woman came to our observation for the presence of an asymptomatic pulsatile mass that had arisen in her left leg 6 months earlier. Her medical history showed hypertension and a previous (33 years before) massive hemorrhage during childbirth that lead to the bilateral ligation of hypogastric arteries. No trauma or infection was recorded. On medical examination the patient presented with a pulsatile mass on the lateral face of the distal left leg portion, about 10 cm in length; left leg diameter at mass level was 2 cm greater than right leg diameter; peripheral pulse was normal at left ankle, while at right ankle only posterior tibial artery pulse was present. Duplex scan showed a giant aneurysm of the ATA (maximum diameter 4 cm) and patency of the infrapopliteal vessels. Division of Vascular and Endovascular Surgery, University of Genoa, IRCCS San Martino IST, Genova, Italy.

Abdominal and lower limb computed tomography angiography showed a dilation of the proximal ATA, expanding into an aneurysm in the middle third of ATA. Maximum diameter was 4.1 cm and length was 10 cm, with partial wall thrombosis in the proximal portion; no other aneurysms were found and the infrapopliteal arteries were patent (Fig. 1). The patient, under subarachnoid anesthesia, underwent intraoperative angiography, through common femoral artery percutaneous access, that showed patency of the proximal ATA and occlusion of its postaneurysmal part. Therefore we decided to perform aneurysmectomy and surgical arterial ligation through a lateral access. We used a 6 mm balloon in the proximal ATA to control arterial inflow. No backflow from the distal tibial artery was found (Fig. 2). The aneurysmal sac, in the absence of signs of dystrophy, was sutured and a sample was sent to laboratory for further analysis and storage in Vascular Biobank. The final angiographic examination showed patency of the 2 remaining vessels and no leakage from the sutured ATA. The distal ATA was perfused with a backflow from the posterior tibial artery. Five days later the patient was discharged without any sign of ischemia. A 6-month follow-up with ultrasound examination showed patency of all remaining vessels, while the patient remained pain free with no sign of ischemia. Histological analysis revealed disorganization of the aneurysmal tissue, inflammatory cell recruitment, and elastin fiber degradation in both samples (Fig. 3).

Correspondence to: Giovanni Spinella, MD, Division of Vascular and Endovascular Surgery, University of Genoa, IRCCS San Martino IST, Largo Rosanna Benzi 10, Genova, Italy; E-mail: [email protected]

DISCUSSION

Ann Vasc Surg 2015; 29: 1319.e5–1319.e9 http://dx.doi.org/10.1016/j.avsg.2015.02.028 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: October 26, 2014; manuscript accepted: February 24, 2015; published online: June 16, 2015.

True infrapopliteal aneurysms are a very rare condition. Their most common causes are infection, collagen vascular pathology, or fibromuscular disease, while trauma and iatrogenic injury often 1319.e5

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Fig. 1. Volume rendering series of the aneurysm: (A) lateral view of ATA with the aneurysm; (B) section of the aneurysm at its maximum diameter; (C) ATA and tibioperoneal trunk, and (D) ATA aneurysm in frontal view.

Fig. 2. (A) Difference between the 2 legs; (B) + (C) aneurysmal exposure and evidence of compression on nearby structures.

cause false infrapopliteal aneurysms.6e10 Cases reported in the literature show the rarity of this disease and, according to different reviews, the ATA is the localization of 2e19 reported true aneurysm cases (Table I). Moreover, the most frequent localization is the distal portion of ATA, and sometimes even the dorsalis pedis artery (DPA),10e12 while aneurysms of the proximal ATA are extremely rare: only 2 have been reported in the literature so far.

Conversely, cases caused by trauma or by local infection, which are usually related to bone fractures, inversion injuries, and trauma surgeries, are more commonly located in the distal portion of ATA or in DPA. However, in the available literature, even collagen disease-related ATA aneurysms are usually not found in the proximal ATA tract.6,18 True infrapopliteal aneurysms may be asymptomatic or symptomatic. The symptoms include

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Fig. 3. Evidence of differences between collagen fibers in classical atheromatosis (abdominal aortic aneurysm) and in our case (ATA).

pain or paresthesia because of nerve compression of the anterior, lateral, and posterior compartment of the leg, depending on which infrapopliteal artery is affected by the aneurysm17; partial or total ischemia of the leg caused by distal embolism or occlusion of aneurysm; and rupture of the artery and subsequent hemorrhage and ischemia of the compartment below the aneurysm. Asymptomatic aneurysms usually present themselves as pulsatile masses in the leg.5,16,17

As mentioned above, only a few cases of true infrapopliteal aneurysms are reported in the literature and the decision to treat or not to treat the aneurysm depends on its size and on the presence or absence of symptoms. If the aneurysm is small and asymptomatic, an ultrasound follow-up can be scheduled18; if the aneurysm is large and/or symptomatic, literature data suggest that the best course of action is to treat it.1,4 According to literature data, the best therapeutic choice is autologous

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Table I. Infrapopliteal aneurysm in literature Authors

Aneurysmatic artery Treatment

Barbano et al.19 Faccenna et al.2 Sigterman et al.3 Tshomba et al.4

TP ATA ATA TP

Ferrero et al.5 Hagspiel et al.6

TP TP

Sugimoto et al.7 Kanaoka and Matsuura8 Ferrero et al.9

TPT TP

Cappendijk and Mouthaan11 M€ onig et al.15 Matsushima and Takara16 Shah et al.18 Toncev et al.17 Joglar et al.10 Gabrielli et al.12 Lynch et al.13 Robaldo et al.14

TPT

P

TPT TP TPT TP TP DP DP DP

Endovascular graft Vein graft Vein graft Direct and to end anastomosis Ligation Endovascular embolization Ligation Vein graft Endovascular embolization Vein graft PTFE graft + vein patch Endovascular embolization No treatment Ligation Endovascular graft Ligation Ligation Vein graft

DP, dorsalis pedis; P, peroneal artery; PTFE, polytetrafluoroethylene; TP, posterior tibial artery; TPT, tibioperoneal trunk.

saphenous vein graft, which maintains blood flow continuity and minimizes the risk of peripheral ischemia. Joglar et al. describe a series of 4 cases of infrapopliteal artery aneurysms treated endovascularly with stents approved for coronary intervention, because there are no stents designed for infrapopliteal use. In our case, as the diameter was quite large, we did not opt for this technique.9 When the other vessels are patent, or there is an adequate collateral circulation, or if dystrophy of the arterial wall may jeopardize the results of the graft, artery ligation is described in the literature as a possible treatment, and can be performed by open or endovascular surgery.10 Open ligature is recommended, because by opening the aneurysmal sac, the mass is reduced, but for high-risk patients endovascular ligation treatment (thrombin glue, coil embolization) is a good alternative to surgery.16 In our case, we chose open treatment based on the fact that our patient was considered at very low risk for surgery (no cardiac, pulmonary, or other diseases), great saphenous vein graft was feasible, and mass reduction was necessary to eliminate the difference in size between the legs as well as the

risk of nerve compression. The final decision to ligate the ATA was because of the lack of backflow and of angiographically visible distal ATA; with 2 infrapopliteal vessels out of 3, the patient was already hemodynamically balanced. Histological analysis showed that collagen fibers were sparse and dystrophic, possibly because of a collagen disease: they were different from fibers derived from abdominal aortic aneurysm, whose origin was purely atherosclerotic, and this difference can be explained with collagen disease. A similar case was described by Arici et al.da young woman with bowel and uterine rupture during pregnancy and a spontaneous rupture of a posterior tibial artery pseudoaneurysm a couple of years later, who was diagnosed with EhlerseDanlos syndrome type 4.20 In our case, the patient showed no cutaneous or articular symptoms, and Ehlerse Danlos syndrome type 4 was excluded, but recent literature reports cases of mutations that concern only arterial vessel collagen, and may cause frailty of arterial wall.21 We did not perform genetic testing to ascertain which of the many possible collagen mutations the patient carried, because under Italian law this would have been ethically and economically justified only in the case of a threat to survival of any living relative (siblings, offspring). We opted for a frequent follow-up, with Duplex scan performed every 6 months to verify infrapopliteal vessel patency, and clinical examination to ascertain the absence of pulsating masses in the site of previous surgery and elsewhere.

CONCLUSION The patient’s clinical background does not show any significant disease, trauma, or infection that may be related to this aneurysm. A previous diagnosis of bilateral rupture of hypogastric arteries is the only interesting bit of information that may point to a collagen vascular disorder. We treated the aneurysm with excision and ligation because the outflow was already absent and the patient was asymptomatic. The patient remained asymptomatic for limb ischemia even after surgery, demonstrating that a good collateral blood flow was already established. At the moment of the diagnosis the patient was clear from other aneurysms, and a close follow-up could ensure that, should another aneurysm manifest itself, it would be treated before it could become symptomatic or dangerous for the patient’s health. REFERENCES 1. John SM, Greer MS. True anterior tibial artery aneurysm in a runner. Am Surg 2014;80:E298e9.

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2. Faccenna F, Alunno A, Felli MM, et al. Tibioperoneal true aneurysm: case report and literature review. G Chir 2011;32:379e83. 3. Sigterman TA, Dolmans DE, Welten RJ, et al. Anterior tibial artery aneurysm: case report and literature review. Int J Surg Case Rep 2013;4:243e5. 4. Tshomba Y, Papa M, Marone EM, et al. A true posterior tibial artery aneurysm: a case report. Vasc Endovascular Surg 2006;40:243e9. 5. Ferrero E, Ferri M, Viazzo A, et al. Rupture of a true giant aneurysm of the posterior tibial artery: a huge size of 6 cm on diameter. Ann Vasc Surg 2010;24:1134.e9e13. 6. Hagspiel KD, Bonatti H, Sabri S, et al. Metachronous bilateral posterior tibial artery aneurysms in Ehlers-Danlos syndrome type IV. Cardiovasc Intervent Radiol 2011;34: 413e8. 7. Sugimoto M, Komori K. Mycotic aneurysm of the tibioperoneal trunk which precipitated acute compartment syndrome: report of a case. Surg Today 2012;42:1001e4. 8. Kanaoka T, Matsuura H. A true aneurysm of the posterior tibial artery: a case report. Ann Thorac Cardiovasc Surg 2004;10:317e8. 9. Ferrero E, Ferri M, Carbonatto P, et al. Symptomatic aneurysm of a perforating peroneal artery after a blunt trauma. Ann Vasc Surg 2012;26:277.e1e3. 10. Joglar F, Kabutey NK, Maree A, et al. The role of stent grafts in the management of traumatic tibial artery pseudoaneurysms: case report and review of the literature. Vasc Endovascular Surg 2010;44:407e9. 11. Cappendijk VC, Mouthaan PJ. A true aneurysm of the tibioperoneal trunk. Case report and literature review. Eur J Vasc Endovasc Surg 1999;18:536e7.

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12. Gabrielli R, Rosati MS, Marcuccio L, et al. Mycotic aneurysm of dorsalis pedis artery due to recurrent Candida albicans foot infection. J Vasc Surg 2014;59:1707e8. 13. Lynch JE, Winhler KA, Xenos E. Posttraumatic versus Mycotic Dorsalis Pedis Pseudoaneurysm. Int J Angiol 2013; 22:135e6. 14. Robaldo A, Colotto P, Palombo D. True atherosclerotic pedis artery aneurysm. Interact Cardiovasc Thorac Surg 2010;11:216e7. 15. M€ onig SP, Walter M, Sorgatz S, et al. True infrapopliteal artery aneurysms: report of two cases and literature review. J Vasc Surg 1996;24:276e8. 16. Matsushima K, Takara H. Endovascular treatment for a spontaneous rupture of the posterior tibial artery in a patient with EhlerseDanlos syndrome type IV: report of a case. Surg Today 2009;39:523e6. 17. Toncev S, Sretenovic S, Mitrovic S, et al. An unusual case of aneurysmal tibial nerve compression detected in ED. Am J Emerg Med 2013;31:1155.e1e3. 18. Shah S, Fischman A, Marin M, et al. Spontaneous tibioperoneal trunk and anterior tibial artery pseudoaneurysms. Vasc Med 2012;17:164e7. 19. Barbano B, Gigante A, Zaccaria A, et al. True posterior tibial artery aneurysm in a young patient: surgical or endovascular treatment? BMJ Case Rep 2009. 20. Arici V, Corbetta R, Fossati LG, et al. Acute first onset of Ehlerse Danlos syndrome type 4 with spontaneous rupture of posterior tibial artery pseudoaneurysm. Vascular 2013;21:43e5. 21. Malfait F, Symoens S, De Backer J, et al. Three arginine to cysteine substitutions in the pro-alpha (I)-collagen chain cause Ehlers-Danlos syndrome with a propensity to arterial rupture in early adulthood. Hum Mutat 2007;28: 387e95.

A true giant aneurysm of the anterior tibial artery.

Aneurysms of the anterior tibial artery are rare. We will describe a case of a woman with an asymptomatic true aneurysm of the anterior tibial artery...
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