The Natural and Unnatural History of Congenital Aortic Arch Abnormalities Evaluated in an Adult Survival Cohort

Can J Cardiol. 2019 Apr;35(4):438-445. doi: 10.1016/j.cjca.2018.12.004. Epub 2018 Dec 7.

Abstract

Background: This study describes the different types of congenital vascular rings according to their anatomy, symptoms, and age at clinical onset and reports the surgical outcomes.

Methods: A retrospective observational database study was conducted, reviewing the medical charts of 69 adult survivors with a history of a vascular ring, identified from the Dutch Congenital Cor vitia database.

Results: Median age at presentation was 8.5 years (0-53.0 years). Thirty patients (43.5%) had a "left aortic arch with aberrant right subclavian artery," 21 patients (30.4%) a "double aortic arch," and 16 patients (23.2%) a "right aortic arch with aberrant left subclavian artery." The main symptomatology at presentation comprised respiratory symptoms (82.9%). Almost three-quarters of patients were also diagnosed with asthma/bronchial hyperreactivity. Patients with a double aortic arch had more symptoms than patients with a left aortic arch with aberrant right subclavian artery and right aortic arch with aberrant left subclavian artery (P < 0.001), requiring surgery most often (P < 0.001). In patients with childhood onset of symptoms, preoperative spirometry (ie, peak expiratory flows) was more often abnormal as compared with adult patients (P = 0.007). Surgery was performed in 42.0% of all patients at a median age of 17 years (0-63.0 years). Twenty-four (92.3%) of the operated patients showed improvement or complete relief of symptoms shortly after surgery. Of 26 asymptomatic nonoperated patients, 3 patients (11.5%) eventually developed symptoms.

Conclusions: The low incidence of vascular rings, their anatomic heterogeneity, and a wide range of common symptoms often lead to misdiagnosis. Clinical awareness is warranted as a large subset of patients could benefit from surgery, even at an adult age.

Publication types

  • Observational Study

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aorta, Thoracic / abnormalities*
  • Asthma / epidemiology
  • Bronchial Hyperreactivity / epidemiology
  • Cardiovascular Surgical Procedures / statistics & numerical data
  • Chest Pain / epidemiology
  • Child
  • Child, Preschool
  • Cyanosis / epidemiology
  • Deglutition Disorders / epidemiology
  • Dyspnea / epidemiology
  • Female
  • Humans
  • Infant
  • Infant, Newborn
  • Length of Stay / statistics & numerical data
  • Male
  • Middle Aged
  • Netherlands / epidemiology
  • Respiratory Sounds
  • Respiratory Tract Infections / epidemiology
  • Retrospective Studies
  • Spirometry
  • Subclavian Artery / abnormalities*
  • Survivors*
  • Vascular Ring / epidemiology*
  • Vascular Ring / surgery
  • Young Adult