Ivica Vucemilo1, John A Harlock2, Mohammad Qadura3, Mina Guirgis3, Robert N Gowing3, Jacques G Tittley3. 1. Division of Vascular Surgery, Trillium Health Centre, Mississauga, Ontario, Canada. 2. Division of Vascular Surgery, McMaster University, Hamilton, Ontario, Canada. Electronic address: harlocj@mcmaster.ca. 3. Division of Vascular Surgery, McMaster University, Hamilton, Ontario, Canada.
Abstract
INTRODUCTION: An aberrant right subclavian artery (ARSA) with or without an associated Kommerell's diverticulum (KOD) is a rare vascular anomaly. Patients with an ARSA may present with a variety of symptoms, including rupture. Options for repair include open, endovascular, and a hybrid approach, with no clear consensus on which is best because of the rarity of the anomaly. We present 2 cases that underwent hybrid repair and a systematic review of the literature. METHODS: A systematic review of the literature was performed from 2000-2012 searching for patients with ARSA with or without KOD who underwent endovascular repair. Twenty-four articles were identified, including a few case reports and small case series, for a total of 31 patients. A chart review was also performed on 2 patients at our institution who underwent a hybrid repair for symptomatic ARSA. RESULTS: We report the presenting history, management, and follow-up of 2 symptomatic patients with ARSA with associated KOD. Both patients underwent a hybrid approach for repair, with no reported complications. After postoperative imaging and clinical follow-up, both patients remained in good general condition without signs of vascular complications. Our systematic literature review identified 31 reported cases of patients with ARSA who underwent endovascular repair (10 patients without an associated KOD and 21 patients with a KOD). The patients ranged in age from 21-82 years of age (average: 56 years). Of these patients, 17 (55%) were women. The presenting symptoms varied, and some patients had multiple symptoms noted on presentation, including dysphagia, dyspnea, or asymptomatic patients. In those patients with an associated KOD, the average size of the diverticulum was 3.3 cm (range: 2.3-7 cm). A number of operative strategies were used in the reported cases, depending on the presence of absence of an associated KOD. The average reported duration of hospital stay was 5.4 days (range: 1-60 days). Seven patients had postoperative complications (22%). There were 3 mortalities reported (10%). Only 1 of these could be directly related to the surgical procedure. Reported decrease in aneurysm size was between 25-50%, although this was not reported for most patients. Four of 31 patients (13%) had an endoleak (1 type I endoleak, 2 type II endoleaks, and 1 type IV endoleak). The range of reported follow-up varied between 6 weeks and 92 months, with 9 patients having no follow-up reported. CONCLUSION: Hybrid approach to repair of an ARSA with associated KOD appears to be feasible, safe, and effective. Despite the poor quality and heterogeneity of the evidence available in the literature for this rare condition, we believe that this could be the preferred treatment option for an ARSA either with or without KOD.
INTRODUCTION: An aberrant right subclavian artery (ARSA) with or without an associated Kommerell's diverticulum (KOD) is a rare vascular anomaly. Patients with an ARSA may present with a variety of symptoms, including rupture. Options for repair include open, endovascular, and a hybrid approach, with no clear consensus on which is best because of the rarity of the anomaly. We present 2 cases that underwent hybrid repair and a systematic review of the literature. METHODS: A systematic review of the literature was performed from 2000-2012 searching for patients with ARSA with or without KOD who underwent endovascular repair. Twenty-four articles were identified, including a few case reports and small case series, for a total of 31 patients. A chart review was also performed on 2 patients at our institution who underwent a hybrid repair for symptomatic ARSA. RESULTS: We report the presenting history, management, and follow-up of 2 symptomatic patients with ARSA with associated KOD. Both patients underwent a hybrid approach for repair, with no reported complications. After postoperative imaging and clinical follow-up, both patients remained in good general condition without signs of vascular complications. Our systematic literature review identified 31 reported cases of patients with ARSA who underwent endovascular repair (10 patients without an associated KOD and 21 patients with a KOD). The patients ranged in age from 21-82 years of age (average: 56 years). Of these patients, 17 (55%) were women. The presenting symptoms varied, and some patients had multiple symptoms noted on presentation, including dysphagia, dyspnea, or asymptomatic patients. In those patients with an associated KOD, the average size of the diverticulum was 3.3 cm (range: 2.3-7 cm). A number of operative strategies were used in the reported cases, depending on the presence of absence of an associated KOD. The average reported duration of hospital stay was 5.4 days (range: 1-60 days). Seven patients had postoperative complications (22%). There were 3 mortalities reported (10%). Only 1 of these could be directly related to the surgical procedure. Reported decrease in aneurysm size was between 25-50%, although this was not reported for most patients. Four of 31 patients (13%) had an endoleak (1 type I endoleak, 2 type II endoleaks, and 1 type IV endoleak). The range of reported follow-up varied between 6 weeks and 92 months, with 9 patients having no follow-up reported. CONCLUSION: Hybrid approach to repair of an ARSA with associated KOD appears to be feasible, safe, and effective. Despite the poor quality and heterogeneity of the evidence available in the literature for this rare condition, we believe that this could be the preferred treatment option for an ARSA either with or without KOD.