Aortic involvement in relapsing polychondritis: case-based review

Rheumatol Int. 2021 Apr;41(4):827-837. doi: 10.1007/s00296-019-04468-5. Epub 2019 Nov 25.

Abstract

Relapsing polychondritis is a systemic inflammatory disease that mainly affects ears, nose, eyes, joints, and large airway. Relapsing polychondritis may also affect cardiac valves and large vessels with the aorta being most frequently involved. We conducted a systematic literature review to delineate the clinical characteristics, treatment, and outcome of relapsing polychondritis patients with aortic involvement including thoracic and abdominal aorta, aortic valve, and coronary arteries. 113 patients reported in 85 manuscripts were retrieved through the systematic literature search and references of the selected manuscripts. With the addition of a patient from our center, a total of 114 patients were included in the analyses. Aortic vessel involvement was the predominant type of involvement that was identified in 93 (82%) patients, while aortic valve involvement was identified in 41 patients (36%). The median age at aortic involvement was 37 years [IQR: 30-53] with a delay of 5 years [IQR: 1-8] between first relapsing polychondritis symptom and aortic involvement. Nineteen percent of the patients were asymptomatic at the time of aortic involvement diagnosis. The initial treatment was immunosuppressives in 41 patients (56%) and surgery in 28 patients (38%). The mortality ratio was 27% in a 24 month follow-up [IQR: 7.5-54 months]. Aortic dissection or rupture was the most frequent causes of mortality. Concomitant coronary artery involvement suggested a worse outcome. Aortic involvement in relapsing polychondritis is a mortal complication despite medical and surgical treatments. It may be asymptomatic in 19% of the patients which warrants the importance of screening.

Keywords: Aortic aneurysm; Aortic valve; Aortitis; Polychondritis; Vasculitis.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adult
  • Aortic Aneurysm / diagnostic imaging*
  • Aortic Valve / diagnostic imaging
  • Aortitis / diagnostic imaging*
  • Computed Tomography Angiography / methods
  • Cyclophosphamide / administration & dosage
  • Cyclophosphamide / therapeutic use
  • Female
  • Fluorodeoxyglucose F18
  • Humans
  • Infliximab / therapeutic use
  • Methylprednisolone / administration & dosage
  • Methylprednisolone / therapeutic use
  • Polychondritis, Relapsing / complications
  • Polychondritis, Relapsing / diagnosis*
  • Polychondritis, Relapsing / drug therapy
  • Positron Emission Tomography Computed Tomography
  • Prednisolone / administration & dosage
  • Prednisolone / therapeutic use
  • Tomography, X-Ray Computed / methods*
  • Treatment Outcome

Substances

  • Fluorodeoxyglucose F18
  • Cyclophosphamide
  • Prednisolone
  • Infliximab
  • Methylprednisolone