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      Behcet’s disease involved the root of aorta in the treatment with modified Bentall procedure: a case report

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          Abstract

          Background

          Behcet’s disease (BD) is a multisystemic vasculitis of unknown etiology, the incidence of cardiovascular system involvement is rare, about1–5% (Sakane et al., N Engl J Med 341:1284–91, 1999). BD combined with aortic pseudoaneurysm and aortic valve regurgitation is usually need surgical treatment, but there is controversy about which surgical method to choose.

          Case presentation

          We report a case of BD combined with severe aortic valve regurgitation and two giant pseudoaneurysms of the aortic sinus. The patient underwent modified Bentall procedure (MBP) and use oral immunosuppressive as well as corticosteroid strictly, after 8 months follow-up, the patient recovered well.

          Conclusion

          For patient with aortic valve regurgitation and ascend aortic pseudoaneurysm caused by BD, we recommend modified Bentall procedure when rheumatism in a stable period. Corticosteroids and immunosuppressive drugs should be used before and after surgery.

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          Most cited references21

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          Behçet's disease.

          T Sakane, M Takeno, N. Suzuki … (1999)
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            Behcet’s disease: epidemiology, clinical manifestations, and diagnosis

            Fereydoun Davatchi, Mariam Masoumi, Farimah Ashofteh … (2016)
            Behcet's Disease (BD) is classified among vasculitides. The aim of this review was to put together different known reports in order to help the reader to better understand the disease, to avoid the frequent misdiagnosis, and to decide the best treatment. Areas covered: a) Epidemiology: BD is rare, and is seen along the Silk Road, from 20 to 420/100,000 in Turkey and 80/100,000 in Iran, to 0.64/100,000 in the UK. b) Clinical manifestations: oral aphthosis is seen in more than 95% of patients, genital aphthosis (60-90%), skin (pseudofolliculitis/erythema nodosum, 40-90%), eyes (uveitis/retinal vasculitis, 45-90%), gastrointestinal (diarrhea/hemorrhage/perforation/pain, 4-38%), vascular (venous/arterial thrombosis, aneurysm, 2.2-50%), neurological (all kinds, especially meningo-encephalitis, 2.3-38.5%), and articular (arthralgia/arthritis/ankylosing spondylitis, 11.6-93%). c) Pathergy test is positive in some patients: 8.6% (in India) to 70.7% (in China). This data was extracted from the five nationwide surveys and the largest case series from BD conference reports and a Pubmed search. Expert commentary: Diagnosis is clinical but classification/diagnosis criteria may help. The best criteria for BD is the International Criteria for Behcet's Disease (ICBD). BD is a multisystem disease progressing by attacks and remissions. Each attack may resemble the preceding or it may be different in duration, severity, and the systems involved.
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              Mortality in Behçet's disease.

              K Desseaux, Bertrand Wechsler, M Resche Rigon … (2010)
              To report the long-term mortality in patients with Behçet's disease (BD). A cohort of 817 patients fulfilling the international criteria for BD from a single center in France were analyzed for causes of death, the standardized mortality ratio (SMR), and the factors associated with mortality. Among the 817 patients with BD, 41 (5%) died after a median followup of 7.7 years, of whom 95.1% were male. The mean ± SD age at death was 34.8 ± 11.9 years. Main causes of death included major vessel disease (mainly, arterial aneurysm and Budd-Chiari syndrome) (43.9%), cancer and malignant hemopathy (14.6%), central nervous system involvement (12.2%), and sepsis (12.2%). The mortality rate at 1 year and 5 years was 1.2% and 3.3%, respectively. There was an increased mortality among patients ages 15-24 years (SMR 2.99, 95% confidence interval [95% CI] 1.54-5.39) and those ages 25-34 years (SMR 2.90, 95% CI 1.80-4.49) as compared with age-and sex-matched healthy controls. The mortality decreased in patients older than age 35 years (SMR 1.23, 95% CI 0.75-1.92). In multivariate analyses, male sex (hazard ratio [HR] 4.94, 95% CI 1.53-16.43), arterial involvement (HR 2.51, 95% CI 1.07-5.90), and a high number of BD flares (HR 2.37, 95% CI 1.09-5.14) were independently associated with the risk of mortality. The overall mortality in our BD cohort was 5% after a median followup of 7.7 years. Male sex, arterial involvement, and the number of flares were associated with mortality in BD.
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                Author and article information

                Contributors
                Yingqiang Guo: drguoyq@hotmail.com
                Journal
                Journal ID (nlm-ta): J Cardiothorac Surg
                Journal ID (iso-abbrev): J Cardiothorac Surg
                Title: Journal of Cardiothoracic Surgery
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1749-8090
                Publication date (Electronic): 29 January 2020
                Publication date PMC-release: 29 January 2020
                Publication date Collection: 2020
                Volume: 15
                Electronic Location Identifier: 30
                Affiliations
                ISNI 0000 0001 0807 1581, GRID grid.13291.38, Department of Cardiovascular Surgery, West China Hospital, , Sichuan University, ; Chengdu, Sichuan 610041 People’s Republic of China
                Article
                Publisher ID: 1070
                DOI: 10.1186/s13019-020-1070-0
                PMC ID: 6990569
                PubMed ID: 31996248
                SO-VID: 9aadc1d0-b431-4899-b674-b6e1f398bb04
                Copyright © © The Author(s). 2020
                License:

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Date received : 5 November 2019
                Date accepted : 7 January 2020
                Funding
                Funded by: FundRef http://dx.doi.org/10.13039/501100001809, National Natural Science Foundation of China;
                Award ID: 81470481
                Award Recipient :
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2020

                ScienceOpen disciplines: Surgery
                Keywords: behcet’s disease,aortic valve regurgitation,aortic sinus pseudoaneurysm,bentall procedure
                Data availability:
                ScienceOpen disciplines: Surgery
                Keywords: behcet’s disease, aortic valve regurgitation, aortic sinus pseudoaneurysm, bentall procedure

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