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World Journal of Cardiology World Journal of W J C Cardiology Submit a Manuscript: http://www.wjgnet.com/esps/ World J Cardiol 2016 August 26; 8(8): 488-495 Help Desk: http://www.wjgnet.com/esps/helpdesk.aspx ISSN 1949-8462 (online) DOI: 10.4330/wjc.v8.i8.488 © 2016 Baishideng Publishing Group Inc. All rights reserved. SYSTEMATIC REVIEW Acquired aortocameral fistula occurring late after infective endocarditis: An emblematic case and review of 38 reported cases Salah AM Said, Massimo A Mariani Salah AM Said, Department of Cardiology, Hospital Group Abstract Twente, 7555 DL Hengelo, The Netherlands AIM Massimo A Mariani, Thorax Centre, Department of Cardiac To delineate the features and current therapeutic option Surgery, University Medical Center Groningen, 9713 GZ Gro­ of congenital and acquired aortocameral fistulas (ACF) ningen, The Netherlands secondary to iatrogenic or infectious disorders. Author contributions: Said SAM and Mariani MA contributed METHODS equally to the manuscript. From a PubMed search using the term "aortocameral fistula", 30 suitable papers for the current review were Conflict-of-interest statement: Authors have no conflict of interest in connection with the submitted manuscript. retrieved. Reviews, case series and case reports published in English were considered. Abstracts and reports Data sharing statement: Technical appendix, statistical code, from scientific meetings were not included. A total of and dataset available from the corresponding author at salah. 38 reviewed subjects were collected and analyzed. In [email protected]. Informed consent, verbal, was obtained for addition, another case - an adult male who presented data sharing but the presented data are anonymized and risk of with ACF between commissures of the right and non- identification is negligible. coronary sinuses and right atrium as a late complication of Staphylococcus aureus infective endocarditis of the AV Open-Access: This article is an open­access article which was selected by an in­house editor and fully peer­reviewed by external - is added, the world literature is briefly reviewed. reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY­NC 4.0) license, RESULTS which permits others to distribute, remix, adapt, build upon this A total of thirty-eight subjects producing 39 fistulas were work non­commercially, and license their derivative works on reviewed, analyzed and stratified into either congenital different terms, provided the original work is properly cited and (47%) or acquired (53%) according to their etiology. the use is non­commercial. See: http://creativecommons.org/ Of all subjects, 11% were asymptomatic and 89% licenses/by­nc/4.0/ were symptomatic with dyspnea (21 ×) as the most common presentation. Diagnosis was established by a Manuscript source: Invited manuscript multidiagnostic approach in 23 (60%), single method in Correspondence to: Salah AM Said, MD, PhD, FESC, Depa­ 14 (37%) (echocardiography in 12 and catheterization in rtment of Cardiology, Hospital Group Twente, Geerdinksweg 141, 2), and at autopsy in 2 (3%) of the subjects. Treatment 7555 DL Hengelo, options included percutaneous transcatheter closure in The Netherlands. [email protected] 12 (30%) with the deployment of the Amplatzer duct or Telephone: +31­88­7085286 septal occluder and Gianturco coil and surgical correction Fax: +31­88­7085289 in 24 (63%). Received: February 13, 2016 CONCLUSION Peer-review started: February 16, 2016 Acquired ACF is an infrequent entity which may occur First decision: April 15, 2016 Revised: May 5, 2016 late after an episode of endocarditis of the native AV. The Accepted: June 27, 2016 management of ACF is generally by surgical correction Article in press: June 29, 2016 but non-surgical device intervention has recently been Published online: August 26, 2016 introduced as a safe alternative. WJC|www.wjgnet.com 488 August 26, 2016|Volume 8|Issue 8| Said SAM et al . Acquired aortocameral fistula Key words: Aortic-atrial shunt; Aortic-atrial fistulas; Infective Abstracts and reports from scientific meetings were not endocarditis; Late complication; Surgical correction included. From 30 publications, 38 reviewed subjects were collected and analyzed. Data were analyzed using © The Author(s) 2016. Published by Baishideng Publishing descriptive statistics. Group Inc. All rights reserved. Statistical analysis Core tip: Aortocameral fistula is an uncommon comp- In contrast to classic meta­analysis, the outcome is lication of native aortic valve (AV) endocarditis, which is defined here as the percentages of an event (without associated with high morbidity and mortality. Acquired comparison) in observed patients. aortocameral fistulas (ACF) may originate from any of the three sinuses of Valsalva. Audible continuous murmur may raise suspicion for the presence of ACF. Congenital Additional clinical case fistulas are less commonly reported than the acquired An adult male presented with ACF between the types. Acquired ACF may occur late after an episode of junction of RCS­NCS and RA as a late complication of endocarditis of the native AV. The management of ACF is Staphylococcus aureus infective endocarditis (IE) of the generally by surgical correction but non-surgical device native AV, is added. intervention has recently been introduced as a safe A 44­year­old male survivor of a prior episode of alternative. Another case is added and the world literature Staph­ylococcus aureus IE of the native AV (1998) is briefly reviewed. presented with a recent history of rapid fatigability (2008) during sporting activities. He was afebrile and a continuous murmur was heard. Laboratory results and Said SAM, Mariani MA. Acquired aortocameral fistula occurring chest X­ray were normal. Resting ECG depicted sinus late after infective endocarditis: An emblematic case and review rhythm with signs of left ventricular hypertrophy (LVH). of 38 reported cases. World J Cardiol 2016; 8(8): 488­495 Two­dimensional transthoracic Doppler echocardiography Available from: URL: http://www.wjgnet.com/1949­8462/full/ revealed mild LVH, the right ventricle (RV) was dil­ v8/i8/488.htm DOI: http://dx.doi.org/10.4330/wjc.v8.i8.488 ated and normokinetic, and the tricuspid AV had no vegetation. Color flow mapping revealed evidence of a high velocity shunt between the commissures of the right coronary sinus (RCS) and non­coronary sinuses INTRODUCTION (NCS) terminating into the right atrium (RA) (Figure 1, Supplementary material online, Video 1). Cardiac [1] Aortocameral fistulas (ACF) may be congenital or catheterization demonstrated a shunt between the aorta [2] acquired complicating acute aortic dissection following and the RA and normal left ventricular kinetics (Figure 2, an intimal tear in the vicinity and proximity of the aortic Supplementary material online, Video 2). Hemodynamic [3] root or after aortic valve (AV) replacement . ACF is an evaluation revealed a significant left-to-right shunt (Qp: uncommon complication of native AV endocarditis, which Qs = 2.0:1.0) with normal pulmonary vascular resi- is associated with high morbidity and mortality. ACF stance, normal intracardiac pressures and high resting may originate from any of the three sinuses of Valsalva. cardiac output of 10 L/min. Computed tomography and Audible continuous murmur may raise suspicion for cardiovascular magnetic resonance were not available at [4] the presence of ACF . The clinical manifestations of that time. The fistula was surgically closed (2008). The [2,5] [6,7] ACF may include exertional dyspnea , chest pain , fistula was surgically closed (2008). After establishing [6,8] [9,10] palpitation , congestive heart failure and recurrent median sternotomy, extracorporeal circulation was [11,12] respiratory tract infection . ACF may incidentally performed through standard cannulation of the aorta and [13] be found during routine preoperative examination . right atrium. The heart was arrested with antegrade and Untreated ACF may cause significant morbidity and early selective blood cardioplegia. On inspection, no infectious mortality. The surgical correction of ACF is the treatment masses or evidence of abscess or vegetations were vi­ of choice but percutaneous transcatheter device sible. Further inspection revealed that the ascending intervention has recently been successfully introduced for aorta was not dilated or calcified and the LV showed [5,6,8,9] the closure of ACF . Acquired ACF is an infrequent moderate hypertrophy. After aortotomy, the AV could entity which may occur late after an episode of endo­ be inspected, which was tricuspid with mild thickening carditis of the native AV. Another case of our own is and the fistula was clearly visible between the RCS and added and the world literature is briefly reviewed. NCS terminating into RA. The fistula was closed with 4.0 prolene suture and pledgets. The patient could easily be weaned off after an uneventful procedure. Postoperative MATERIALS AND METHODS transesophageal echocardiography revealed no rest Literature search shunt flow. The patient had an uneventful postoperative From the PubMed search using the term "aortocameral course. The patient had uneventful postoperative course fistula", 30 suitable papers for the current review and regained his non­professional sporting activities were retrieved (Table 1). Reviews, case series and without any limitations. After 8 years of follow­up, he case reports published in English were considered. remains free of symptoms. The fistula was closed by 4.0 WJC|www.wjgnet.com
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