A case of a 26-year-old man without continuity of blood flow in the level of the isthmus of the
aorta is described. During interventional catheterization, we reconstructed this continuity and
implanted a vascular stent. (Cardiol J 2008; 15: 80-84

Białkowski, Jacek

Kusa, Jacek

Szkutnik, Małgorzata

English

Via Medica Journals

CASE REPORTCardiology Journal2008, Vol. 15, No. 1, pp. 80–84Copyright © 2008 Via MedicaISSN 1897–559380 www.cardiologyjournal.orgAddress for correspondence: Dr n. med. Jacek KusaClinic of Congenital Heart Disease and Pediatric CardiologySilesian Centre for Heart Disease, ZabrzeSzpitalna 2, 41–800 Zabrze, PolandTel./fax: +48 32 271 34 01; e-mail: jkusa@poczta.onet.plReceived: 23.05.2007 Accepted: 21.11.2007Percutaneous reconstruction of the continuity ofa functionally interrupted aortic arch using a stentJacek Kusa, Małgorzata Szkutnik and Jacek BiałkowskiDivision of Congenital Heart Disease and Pediatric Cardiology,Silesian Centre for Heart Disease, Zabrze, PolandAbstractA case of a 26-year-old man without continuity of blood flow in the level of the isthmus of theaorta is described. During interventional catheterization, we reconstructed this continuity andimplanted a vascular stent. (Cardiol J 2008; 15: 80–84)Key words: aortic coarctation, stentIntroductionCoarctation of the aorta (CoA) is a commoncongenital cardiovascular defect [1]. It is usuallydiagnosed in children but occasionally in adults. Thelatter patients are usually asymptomatic, and thediagnosis of CoA is made during an attempt to iden-tify the cause of high blood pressure. Untreated CoAmost commonly leads to death by the fifth decadeof life [2, 3]. Early symptoms include intermittentclaudication, headaches, vomiting, nausea and diz-ziness. Less common clinical manifestations havealso been reported, including congestive heart fail-ure, infectious endocarditis involving aortic intima,true ascending or descending aortic aneurysm, aor-tic dissection and cerebrovascular events [3, 4].Surgical treatment of CoA was described for thefirst time in 1945, with a number of methods of sur-gical repair of the aorta introduced since that time.In 1979 a postmortem study showed the feasibilityof balloon angioplasty of lesions, and the first per-cutaneous balloon angioplasty of CoA in an infantwith heart failure was performed in 1982. Soon thisprocedure was also introduced in adult patients. Dueto suboptimal results caused by vessel hypoplasia,elastic recoil or restenosis, modifications of balloonangioplasty were sought to solve these problems. In1993 the first stent implantation into the aortic isth-mus was performed in clinical practice by Suares deLezo (followed by the publication of these results in1995) [2, 5, 6], and since that time this method hasbecome widely used in all age groups [4, 7–10].In this report, we present a patient with criti-cal CoA, also known as functionally [11] or physio-logically interrupted aortic arch, with coexistingatrial septal defect who was treated with percuta-neous stent implantation into the aortic isthmus.Case reportA 26-year-old man, a foreigner, was presentedto our centre for percutaneous treatment of CoA.The defect was diagnosed in childhood, but the par-ents of the patient did not consent to the suggest-ed surgical treatment. The patient grew and devel-oped normally during childhood, but he requiredtreatment for his hypertension. For six years bloodpressure was well controlled using two antihyper-tensive drugs. In addition, intermittent claudicationdeveloped, leading to progressively more severesymptoms.On admission, the patient was in a good generalclinical condition. No pulses within the lower limbswere noted during the physical examination, andheart auscultation revealed fixed splitting of thesecond heart sound and late systolic murmur in the81 Jacek Kusa et al., Reconstruction of the continuity of the aortic archwww.cardiologyjournal.orgleft interscapular area and in the 2nd–3rd left inter-costal space. Blood pressure on the upper limbs was155/95 mm Hg. Chest radiography showed normalcardiac silhouette, abnormal contour of the aorta andrib notching. Echocardiography revealed morpho-logically severe stenosis of the aortic isthmus witha pressure gradient that was difficult to measure,and slow flow in the abdominal aorta. Cardiac cham-ber dimensions were normal for body mass, and thethickness of left ventricular myocardium was in-creased by 22%. In addition, bileaflet aortic valveand an atrial septal defect with a diameter of 12 mmand a left-to-right shunt were found. The patientwas scheduled for an attempt at percutaneous dila-tation of CoA. Simultaneous closure of the atrialseptal defect was also contemplated but the patientdid not agree to the latter due to financial reasons.Cardiac catheterization was performed followingintravenous sedation using midazolam and morphine,under antibiotic coverage (cephazolin), by percutane-ous femoral artery approach using 6 F sheath. A mul-tipurpose catheter was introduced into the descend-ing aorta, and multiple attempts to cross the coarcta-tion were made using various guidewires (Teflon,hydrophilic and coronary), but the catheter only en-tered numerous wide collateral vessels (Fig. 1).The multipurpose catheter was replaced with a 6 Fpigtail catheter used for aortography below the aor-tic isthmus but no uncontrasted blood flow to thedescending arm of the aortic arch was seen (Fig. 2).The aortic diameter at the level of the diaphragmwas 21 mm. Interruption of the aortic arch was thensuspected and a 6 F multipurpose catheter was in-troduced through the right femoral vein (via a 6 Fsheath) to the left atrium, left ventricle and the as-cending aorta. Using a 260 cm-long 0.035-inchguidewire, the multipurpose catheter was replacedwith a pigtail catheter used for aortography of theascending aorta. Again, no flow through the entirelength of the aorta could be seen (Fig. 3), and thedescending aorta was visualized after a delay nec-essary for the inflow of contrasted blood throughcollateral vessels. Anatomic continuity of aorticwalls was found, with the vessel lumen not seen(atresia? septating membrane?) at a length of only1 mm (14 mm below the origin of the left subclavi-an artery) and a clearly visible cone of blind-endedproximal aorta. During multiple attempts to crossthe presumed CoA, a 0.035-inch hydrophilic Teru-mo guidewire entered the descending aorta (perfo-ration of an aortic septating membrane? crossingnarrow minimal CoA lumen that was not seen inFigure 1. Collateral vessels.82Cardiology Journal 2008, Vol. 15, No. 1www.cardiologyjournal.orgaortography?). Using this guidewire, a multipurposecatheter was introduced into the descending aortaand the guidewire was replaced with a 260 cm-long0.035-inch Teflon guidewire, with the tip of thelatter left in the abdominal aorta. The pressuregradient between the ascending and descendingaorta was estimated at 61 mm Hg. Subsequently,a Microvena snare catheter advanced through thefemoral artery sheath was used to catch and exte-riorize the guidewire tip. Using this arteriovenousloop, and after replacing the femoral artery sheathwith a long 12 F Mullins sheath, a 16 mm × 4 cmMaxi balloon with mounted P 4014 stent (Johnson& Johnson) was introduced into CoA. The stent wasexpanded just below the origin of the left subclavi-an artery, resulting in dilatation of the stenosedaortic segment to 16 mm (Fig. 4). Following theprocedure, the pressure gradient decreased to zero.Follow-up aortography showed a minimal aneurysmand incomplete apposition of the stent to the aorticwalls above and below the site of previous steno-sis. A follow-up catheterization with a view to re-peated stent dilatation was scheduled for six monthslater. The patient presented for the follow-up eval-uation 18 months later. During that time he had nosymptoms, and antihypertensive drugs were initiallyFigure 2. Aortography below the isthmus does notreveal ‘negative’ contrast.Figure 4. Aortography after stent implantation.Figure 3. Aortography reveals stopped blood flow inthe isthmus of the aorta.83 Jacek Kusa et al., Reconstruction of the continuity of the aortic archwww.cardiologyjournal.orgused in reduced doses and later completely with-drawn. Blood pressure was 120/80 to 135/85 mm Hg.Physical examination showed normal pulses inthe lower limbs, and echocardiography revealednormal flow in the abdominal aorta with a mini-mal diastolic plateau. Pressure gradient at theaortic isthmus was estimated at 20 mm Hg. Theatrial septal defect size decreased from 12 to7 mm. The patient did not give consent for stentdilatation.DiscussionCoarctation of the aorta may be treated surgi-cally or percutaneously. The choice of the therapeu-tic option should be based on patient preference.This patient presented to our centre for percuta-neous treatment.Diagnostic evaluation revealed an extremeform of CoA that occurs very rarely in this age groupand is the functional equivalent of an interruptedaortic arch. Nevertheless, we decided to rise to thechallenge and attempt percutaneous restoration ofthe aortic lumen [12] using a transvenous approachto the ascending aorta (through the atrial septaldefect).Having the choice between balloon angioplastyand percutaneous stent implantation, we decided toperform the latter. Conventional indications forstenting include unfavourable anatomy for balloonangioplasty, such as tubular coarctation, hypoplasiaof the isthmus, long stenosed aortic segment [2, 8]and the lack of the effect of balloon angioplasty orrestenosis following angioplasty. However, stentsare now used more commonly and in some centresthey have become a first line treatment in all adultpatients, and in some centres stents are used asa palliative treatment even in small children [5]. Lit-erature review suggests that the advantages ofstents compared to balloon angioplasty include sig-nificantly lower residual pressure gradient andlower rates of restenosis and direct aortic wall dam-age [5, 10]. According to Suarez de Lezo et al. [5],even a long time after balloon angioplasty there isa 10 to 15 percent risk of the development of an an-eurysm. Data are inconsistent in regard to the rateof aneurysm development following both types ofpercutaneous treatment: some authors suggest thatthis occurs at a similar rate following both stentingand balloon angioplasty, estimated at 6–7%, but oth-er investigators reported more frequent aneurysmdevelopment following balloon angioplasty [5, 6, 13].Aneurysms develop due to damage of the aortic me-dia, and overestimation of the diameter of the bal-loon used for angioplasty is suggested as a contrib-uting cause [6]. In some cases, stents may protectagainst such complications, so the rate of aneurysmdevelopment might be lower following stenting [13].Appropriate sizing of the balloon is very important.The balloon diameter should be 1–2 mm smallerthan the diameter of the aortic isthmus (most com-monly at the level of the origin of the left subclavi-an artery), and it should not be larger that the di-ameter of the aorta at the level of the diaphragm[6, 14]. If an aneurysm develops, it might be occlud-ed percutaneously using coils or a covered stent[7, 9, 15]. Stent implantation may also result in othercomplications including stent fracture or migration,thromboembolic events or aortic rupture [7, 8]. Therisk of the latter may by reduced by the use of anappropriately sized balloon, but on the other hand,the use of a smaller balloon to expand the stent maybe responsible for incomplete apposition of the stentto the aortic wall below the treated segment [10],as was the case in our patient. However, if the ste-nosis is very severe, a two-stage approach is advo-cated, with repeated procedure at approximately sixmonths. During this time, a scar forms in the areaof previous angioplasty, and then stent redilatationis performed using balloons to shape appropriatelythe stent as dictated by the vessel morphology.Another decision is the choice between balloon pre-dilatation and primary stenting. Most of the availa-ble data from the literature suggest that primarystenting reduces the risk of aortic wall damage andthis approach is used in most centres [2, 7, 8]. Inour experience, balloon predilatation is helpful incases of restenosis following surgical treatment asthis approach allows assessment of the aortic wallsusceptibility to expansion, an important issue withhard, incompliant lesions. With this information, wecan avoid implanting a stent that could not be suc-cessfully dilated later.Our patient had critically narrow CoA. Due tothe risk of aortic rupture, we considered implanta-tion of a covered stent. However, this is associatedwith a risk of obstructing blood flow to intercostalarteries supplying the spinal cord [9, 16, 17], so wedecided to implant a regular uncovered stent, hav-ing a covered stent at our disposal in case of anyadverse effects of stenting. During subsequent fol-low-up, the patient’s blood pressure lowered, al-though dilatation of the stenosed aortic isthmus,either surgical or percutaneous, does not alwayslead to blood pressure normalization due to persistingreceptor dysregulation [6]. In fact, this effect is lessoften seen in patients above 4–6 years of age. Availa-ble data suggest that blood pressure normalization84Cardiology Journal 2008, Vol. 15, No. 1www.cardiologyjournal.orgfollowing dilatation of CoA occurs in 50–79% ofpatients [6].Unfortunately, even successful lowering of thepressure gradient does not fully protect the patientfrom increased cardiovascular morbidity duringadult life, although this risk is significantly lower.Potential sequelae include hypertension, aneurysmdevelopment, premature coronary artery disease, in-flammation of the intima and formation of a fistula [4].As a result, such patients require subsequent peri-odic cardiovascular evaluation.Percutaneous intervention performed in ourpatient resulted in a significant reduction of theshunt through the abnormal communication at thelevel of the interatrial septum, driven by reducedleft ventricular pressures, and possibly also reducedleft atrial pressures (due to increased diastolic ven-tricular compliance). As the diameter of the atrialseptal defect decreased by 40%, it did not requireclosure any longer.ConclusionsPercutaneous treatment of CoA is feasible evenin patients with functionally interrupted aortic arch-es or extremely severe forms of CoA. For such pro-cedures to be performed, all necessary equipmentincluding covered stents must be available in thecardiac catheterization laboratory. In addition, imme-diate access to cardiac surgery is required in casesurgical treatment should become necessary.AcknowledgementsThe authors appreciate help of dr Piotr Jędrusikwith preparation of the authorized English versionof the manuscript.Patient provided written consent for perform-ing procedure consisiting of stent placement.The authors do not report any conflict ofinterest regarding this work.References1. Rocchini AP. Coarctation of the aorta and interrupt-ed aortic arch. In: Moller JH, Hoffman JIE ed. Pediat-ric cardiovascular medicine. Churchill Livingstone,New York 2000: 567–593.2. Zabal C, Attie F, Rosas M, Buendia-Hernanadez A,Garcia-Montes JA. The adult patient with native co-arctation of the aorta: balloon angioplasty or primarystenting? Heart, 2003; 89: 77–83.3. Ramnarine I. Role of surgery in the management ofthe adult patient with coarctation of the aorta. Post-grad Med J, 2005; 81: 243–247.4. Oliver JM, Gallego P, Gonzalez A, Arorca A, Bret M,Mesa JM. Risk factors for aortic complications inadults with coarctation of the aorta. J Am Coll Cardiol,2004; 44: 1641–1647.5. Suarez de Lezo J, Pan M, Romero M et al. Percuta-neous interventions on severe coarctation of the aor-ta: a 21-year experience. Pediatr Cardiol, 2005; 26:176–189.6. Fawzy ME, Awad M, Hassan W, Al Kadhi Y, Shoukri M,Fadley F. Long-term outcome (up to 15 years) ofballoon angioplasty of discrete native coarctation ofthe aorta in adolescents and adults. J Am Coll Cardiol,2004; 43: 1062–1067.7. Chessa M, Carrozza M, Butera G et al. Results andmid-long-term follow-up of stent implantation for na-tive and recurrent coarctation of the aorta. Eur Heart J,2005; 26: 2728–2732.8. Thanopoulos BD, Hadjinikolaou L, Konstadopoulou GN,Tsaousis GS, Triposkiadis F, Spirou P. Stent treat-ment for coarctation of the aorta: intermediate termfollow up and technical consideration. Heart, 2000;84: 65–70.9. Kusa J, Bialkowski J, Szkutnik M. Percutaneous im-plantation of stent in 52-year old man with severecoarctation of the aorta and congestive heart failure.Folia Cardiol, 2003; 10: 225–229.10. Harrison DA, McLaughlin PR, Lazzam C, Connelly M,Benson LN. Endovascular stents in the manage-ment of coarctation of the aorta in the adolescentand adult: One-year follow-up. Heart, 2001; 85:561–566.11. Ewert P, Schubert S, Peters B, Abdul-Khaliq H,Nagdyman N, Lange PE. The CP stent-short, long,covered-for treatment of aortic coarctation, steno-sis of pulmonary arteries and caval veins, and Fon-tan anastomosis in children and adults: An evalua-tion of 60 stents in 53 patients. Heart, 2005; 91:948–953.12. Joseph G, Mandalay A, Rajendiran G. Percutane-ous recanalization and balloon angioplasty of con-genital isolated local atresia of the aortic isthmusin adults. Catheter Cardiovasc Inter, 2001; 53:535–541.13. Pedra CA, Fontes VF, Esteves CA et al. Stenting vs.balloon angioplasty for discrete unoperated coarcta-tion of the aorta in adolescents and adults. CatheterCardiovasc Inter, 2005; 64: 495–506.14. Ovaert C, McCrindle BW, Nykanen D, McDonald C,Freedom RM, Benson LN. Balloon angioplasty of na-tive coarctation: clinical outcomes and predictors ofsuccess. J Am Coll Cardiol, 2000; 35: 988–996.15. Cheatham JP. Stenting of coarctation of the aorta.Cathet Cardiovasc Inter, 2001; 54: 112–125.16. Duke C, Qureshi SA. 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Percutaneous reconstruction of the continuity of a functionally interrupted aortic arch using a stent

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Percutaneous reconstruction of the continuity of a functionally interrupted aortic arch using a stent

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