Case report

Aortic aneurysms after subclavian angioplasty repair of coarctation of the aorta

Coarctation of the aorta is a common cardiac disease in Turner syndrome. Evidence indicates that surgery and balloon angioplasty in infants and small children do not have any added risk of mortality or complication in these patients. Stenting in older patients may, however, pose higher risks of arterial wall injury and mortality.

In this case series, we describe 15 patients with coarctation of the aorta in Turner syndrome: 9 received stenting, 4 underwent surgery, and 2 were treated via balloon angioplasty.

Dissection occurred in 2 patients after stenting: 1 in the aorta and the other in the external femoral artery. Both were managed promptly without any mortality or serious damage, one percutaneously and the other surgically.

Awareness of increased risks and preparedness for prompt interventions in case of an acute arterial wall injury are recommended when coarctation stenting is done for a patient with Turner syndrome.

We report our experience with stent implantation for treatment of aortic coarctation in patients with Turner syndrome. Ten consecutive patients with Turner syndrome and aortic coarctation (median age 12 years, range 9 to 24) underwent coarctation stenting. Of these, 6 patients were treated for isolated coarctation and 4 for recurrent coarctation (3 after balloon dilation and 1 after balloon dilation and surgical repair). Bare metal stents were implanted in 7 patients and covered stents in 3. Immediately after stent implantation, peak systolic gradient decreased from 46.1 ± 24.3 to 1.9 ± 2.1 mm Hg (p <0.001). Aortic diameter at coarctation site increased from 5.1 ± 3.2 to 15.3 ± 2.0 mm after stenting (p <0.001). There were no deaths or procedure-related complications. During a median follow-up of 30.5 months, no patient developed restenosis. Two patients developed late aortic aneurysms at the coarctation site. In conclusion, stent implantation for aortic coarctation in patients with Turner syndrome appears to be a safe and effective alternative to surgical repair. Larger cohorts and longer-term follow-up are required to determine the effects of the procedure on the aortic wall.

Although the immediate and intermediate-term results of balloon angioplasty (BA) for patients with aortic coarctation (AC) have been encouraging, there is paucity of data on long-term follow-up results. This study evaluated the long-term (up to 22 years) follow-up results of BA in adolescent and adult patients with discrete (shelf-like) coarctation of the aorta.

Follow-up data of 58 patients (mean age 24 ± 9 years) undergoing BA for discrete AC at median interval of 13.4 years including cardiac catheterization, magnetic resonance imaging, and Doppler echocardiography form the basis of this study.

No early deaths occurred. Balloon angioplasty produced immediate reduction in peak AC gradient from 60 ± 22 mm Hg to 8.5 ± 8 mm Hg (P < .0001). Follow-up catheterization 12 months later revealed a residual gradient of 5 ± 6.4 mm Hg (P = .01). Five patients (8%) with suboptimal initial outcome (peak gradient >20 mm Hg) developed restenosis, and 4 of these had successful repeat angioplasty. Aneurysm developed at the site of dilatation in 4 patients (7%). Magnetic resonance imaging follow-up results revealed no new aneurysm. In one patient, the aneurysm increased in size, but no recoarctation or appreciable changes in the Doppler gradient across the AC site was noted. The blood pressure had normalized without medical treatment in 29 (50%) of the 58 patients.

Long-term results of BA for discrete AC are excellent and should be considered as first option for treatment of this disease.

Presentamos dos casos de aneurisma de aorta torácica descendente (AATD) secundarios a coartación aórtica (CA) tratados mediante técnica endovascular.

Caso 1: varón de 28 años intervenido de CA a los 3 meses de edad (coartectomía con anastomosis terminoterminal) y reintervenido a los 8 años (aortoplastia con parche de Dacron), que presenta una dilatación de 60 mm del área previamente intervenida y que interesa la salida de la arteria subclavia izquierda (ASI), con gradiente transaórtico de 21 mmHg. Requirió la realización de un bypass carotidosubclavio previo a la reparación aneurismática, implantando posteriormente dos endoprótesis solapadas tipo Talent de 26 × 130 mm y de 30 × 36 × 115 mm. Caso 2: mujer de 35 años, sin antecedentes personales de interés, con AAT descendente de 42 × 39 mm a 16 mm de la salida de la ASI, portadora de CA con gradiente transaórtico de 28 mmHg. Se implantó una endoprótesis aórtica tipo Talent de 32 × 130 mm con extremo proximal no recubierto en cayado aórtico proximal a ASI, sin oclusión de la salida de troncos supraaórticos, asociando una dilatación con balón de la zona residual de estenosis de la coartación. En ambos casos los AAT se excluyeron totalmente, sin fugas demostrables angiográfica ni topográficamente y sin gradientes transaórticos residuales. El resultado en el control clínico a 24 y 18 meses, los pacientes se encuentran asintomáticos, con el saco aneurismático trombosado y ausencia de fugas.

Pese a que la respuesta del material y duración a largo plazo de las actuales endoprótesis se desconocen, el procedimiento endovascular puede convertirse en la técnica de elección en un número sustancial de pacientes con patología en aorta torácica descendente.

We report on two cases of aneurysm of the descending thoracic aorta (DTA) secondary to aortic coarctation (AC) which were treated using endovascular techniques.

Case 1: a 28-year-old male who underwent an intervention to correct AC at the age of 3 months (coarctectomy with end-to-end anastomosis) and was submitted to surgery again at the age of 8 years (Dacron patch aortoplasty); there was a 60 mm dilatation of the area of the previous intervention with involvement of the exit point of the left subclavian artery (LSA), with a transaortic gradient of 21 mmHg. The patient required the placement of a carotid-subclavian bypass before aneurysmal repair, and later two overlapping 26 × 130 mm and 30 × 36 × 115 mm Talent-type stent grafts were implanted. Case 2: a 35-year-old female with no history of relevant clinical events, who had a 42 × 39 mm aneurysm of the DTA 16 mm from the exit point of the LSA; the patient was a carrier of AC with a transaortic gradient of 28 mmHg. A 32 × 130 mm Talent-type aortic stent graft with a non-covered proximal end was placed in the aortic arch proximal to the LSA, without occluding the exit of the supra-aortic trunks, and with the association of balloon dilatation of the stenotic region that remained from the coarctation. In both cases the aneurysms of the DTA were totally excluded, with no leaks that could be verified angiographically or topographically, and with no residual transaortic gradients. Results in the clinical control at 24 and 18 months showed that the patients are asymptomatic, with a thrombosed aneurysmal sac and absence of leaks.

Despite the fact that the performance of the materials and the long-term duration of stent grafts in current use are unknown, the endovascular procedure may become the preferred technique in a substantial number of patients with a pathological condition affecting the descending thoracic aorta.

Apresentamos dois casos de aneurisma da aorta torácica descendente (AATD) secundários a coarctação aórtica (CA) tratados mediante técnica endovascular.

Caso 1: homem de 28 anos intervencionado por CA aos 3 meses de idade (coarctectomia com anastomose terminoterminal) e reintervencionado aos 8 anos (aortoplastia com tela de Dacron), que apresenta uma dilatação de 60 mm da área previamente intervencionada e que respeita à saída da artéria subclávia esquerda (ASI), com gradiente transaórtica de 21 mmHg. Requereu a realização de um

carotidosubclávio prévio para a reparação aneurismática, implantando posteriormente duas endopróteses ocultas tipo Talent de 26 × 130 mm e de 30 × 36 × 115 mm. Caso 2: mulher de 35 anos, sem antecedentes pessoais de interesse, com AAT descendente de 42 × 39 mm a 16 mm da saída da ASI, portadora de CA com gradiente transaórtica de 28 mmHg. Implantou-se uma endoprótese aórtica tipo Talent de 32 × 130 mm com extremidade proximal não recoberta no arco aórtico junto à ASI, sem oclusão da saída das artérias supra-aórticas, associando uma dilatação com gases da zona residual de estenose da coarctação. Em ambos os casos, os AAT excluem-se totalmente, sem fugas demonstráveis, angiográfica nem topograficamente, e sem gradiente transaórtica residual. No resultado do controlo clínico a 24 e 18 meses, os doentes encontram-se assintomáticos, com a bolsa aneurismática trombosada e ausência de fugas.

Pesa o facto de a resposta do material e duração a longo prazo das actuais endopróteses se desconhecer, o procedimento endovascular pode converter-se na técnica de eleição num número substancial de doentes com patologia na aorta torácica descendente.