Author:
Lacoste Chloé,Mansencal Nicolas,m'rad Mona Ben,Goulon-Goeau Catherine,Cohen Pascal,Guillevin Loïc,Hanslik Thomas
Abstract
Abstract
Background
Antineutrophil cytoplasmic antibodies (ANCA)-associated systemic vasculitides have a variety of presentations, but cardiac valvular involvement is rarely diagnosed and its management is not established.
Case presentation
We report the case of a 44 year old man who presented with an ANCA-associated systemic vasculitis and aortic regurgitation of unusual mechanism. Transthoracic and transesophageal echocardiography disclosed septal hypertrophy preventing a complete closure of the aortic valve and thus responsible for a massive aortic regurgitation. After 4 months of immunosuppressive therapy, the valve lesion did not subside and the patient had to undergo aortic valve replacement. This report also reviews the 20 cases of systemic ANCA-associated vasculitis with endocardial valvular involvement previously reported in the English language medical literature.
Conclusions
Valvular involvement in ANCA-associated systemic vasculitides is rarely reported. Most of these lesions are due to Wegener's granulomatosis and half are present when the diagnosis of vasculitis is made. The valvular lesion is usually isolated, aortic regurgitation being the most frequent type, and often requires valve replacement in the months that follow it's discovery.
Publisher
Springer Science and Business Media LLC
Subject
Orthopedics and Sports Medicine,Rheumatology
Reference21 articles.
1. Chen M, Yu F, Zhang Y, Zou WZ, Zhao MH, Wang HY: Characteristics of Chinese patients with Wegener's granulomatosis with anti-myeloperoxidase autoantibodies. Kidney Int. 2005, 68 (5): 2225-2229.
2. Anthony DD, Askari AD, Wolpaw T, McComsey G: Wegener granulomatosis simulating bacterial endocarditis. Arch Intern Med. 1999, 159 (15): 1807-1810.
3. Attaran S, Desmond M, Ratnasingham J, Scawn N, Pullan DM: Mitral valve involvement in Wegener's granulomatosis. Ann Thorac Surg. 2010, 90 (3): 996-997.
4. Bruno P, Le Hello C, Massetti M, Babatasi G, Saloux E, Galateau F, Khayat A: Necrotizing granulomata of the aortic valve in Wegener's disease. J Heart Valve Dis. 2000, 9 (5): 633-635.
5. Dabbagh S, Chevalier RL, Sturgill BC: Prolonged anuria and aortic insufficiency in a child with Wegener's granulomatosis. Clin Nephrol. 1982, 17 (3): 155-159.
Cited by
37 articles.
订阅此论文施引文献
订阅此论文施引文献,注册后可以免费订阅5篇论文的施引文献,订阅后可以查看论文全部施引文献