Literature DB >> 19217648

Aortitis and spondyloarthritis--an unusual presentation: case report and review of the literature.

Lihi Eder1, Mouhannad Sadek, Heather McDonald-Blumer, Dafna D Gladman.   

Abstract

OBJECTIVES: To report a patient with aortic regurgitation (AR) and aortitis, who failed to respond to multiple attempts at surgical replacement of the affected valve and adjacent aorta, and in whom a late diagnosis of spondyloarthritis (SpA) was made. The relevant literature on the association of SpA and aortitis is reviewed.
METHODS: Descriptive case report of a patient with AR secondary to aortic dilation and aortitis with a late diagnosis of SpA, and a review of the relevant literature (PubMed search 1956 through April 2008).
RESULTS: A 36-year-old man was admitted to the hospital for replacement of his aortic valve and aortic valve root as a result of AR after a dehiscence of a mechanical prosthetic valve and subsequent aortic pseudoaneurysm. His past medical history included 2 prior aortic valve replacements that failed due to late postoperative dehiscence. Histological findings from the aortic wall after the third operation showed signs of aortitis. A rheumatologic workup revealed a 6-year history of mild low back pain, radiological evidence of bilateral sacroiliitis, and presence of HLA-B27. A diagnosis of SpA, most likely ankylosing spondylitis (AS), was made. On reviewing the literature, AR has been diagnosed in 2 to 12% of the patients with AS, most of whom had long-lasting disease. There have been several reports of uncommon presentations of acute AR in young patients with juvenile AS; however, no similar adult case was found.
CONCLUSION: This unusual case emphasizes the importance of early and adequate workup for SpA in young patients with lone AR or aortic dilation. Copyright 2010 Elsevier Inc. All rights reserved.

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Year:  2009        PMID: 19217648     DOI: 10.1016/j.semarthrit.2008.11.004

Source DB:  PubMed          Journal:  Semin Arthritis Rheum        ISSN: 0049-0172            Impact factor:   5.532


  2 in total

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  2 in total

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