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


CONCLUSIONS
This unusual case emphasizes the importance of early and adequate workup for SpA in young patients with lone AR or aortic dilation.
Eder
Lihi
L
Centre for Prognosis Studies in the Rheumatic Diseases, Toronto Western Hospital, Toronto, Canada. benlihi@gmail.com
Sadek
Mouhannad
M
McDonald-Blumer
Heather
H
Gladman
Dafna D
DD
D000328:Adult; D001022:Aortic Valve Insufficiency / Q000175:diagnosis* / Q000209:etiology; D001025:Aortitis / Q000150:complications / Q000175:diagnosis*; D015796:HLA-B27 Antigen / Q000097:blood; D006801:Humans; D008297:Male; D013167:Spondylitis, Ankylosing / Q000097:blood / Q000150:complications / Q000175:diagnosis*; D014057:Tomography, X-Ray Computed
D015796:HLA-B27 Antigen