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      Aortotracheal fistula after slide tracheoplasty in a patient with dextrocardia, left pulmonary artery sling and tracheal stenosis: a case report

      case-report

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          Abstract

          Background

          Aortotracheal fistula (ATF) is an uncommon and fatal complication of tracheal or aortic surgery, especially among pediatric patients.

          Case presentation

          We reported a case in a 1-year-old boy with dextrocardia, left pulmonary artery sling and long segment tracheal stenosis. He received slide tracheoplasty at 9 months of age and had post-operative refractory granulation at distal trachea status post repeated balloon dilatation and laser vaporization. Episodes of hemoptysis occurred on post-operative day 81. Bronchoscopy revealed a pulsating pseudoaneurysm at lower trachea which ruptured during the procedure

          Urgent surgical repair under cardiopulmonary bypass with deep hypothermic circulatory arrest was done. No recurrent bleeding or significant neurologic deficits noticed at a 4-month follow-up.

          Conclusion

          Congenital anomaly that changes the spatial relationship between trachea and aorta could have contributed to formation of ATF. This warrant future attention when managing tracheal granulation with this not uncommon anatomy.

          Supplementary Information

          The online version contains supplementary material available at 10.1186/s13019-021-01438-6.

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          Most cited references11

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          Postoperative aortic fistulas into the airways: etiology, pathogenesis, presentation, diagnosis, and management.

          Marco Picichè, Ruggero De Paulis, Alessandro Fabbri … (2003)
          Postoperative aortobronchial and aortopulmonary fistulas are rare and late complications of cardiac surgery. They mostly complicate descending thoracic aortic procedures. Hemoptysis is the main symptom, and may be massive or intermittent. The reported interval between the time of operation and the onset of hemoptysis ranges from 3 weeks to 25 years. Diagnostic examinations are often unable to directly visualize a fistula. Indication for surgical or endovascular repair mostly relies on clinical suspicion and nonspecific diagnostic features. Urgent treatment is based on the association of the following elements: (1) hemoptysis, (2) history of previous cardiac or aortic operation, (3) presence of lung infiltrates on the chest roentgenogram, (4) lung hemorrage on the computed tomographic scan, and (5) and visualization of a pseudoaneurysm. Aortobronchopulmonary fistulas are uniformly fatal if untreated. The overall surgical mortality rate is 15.3%. There is no procedure-related mortality after endovascular stent grafting. A review of the English-language literature from 1947 to October 2002 is presented.
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            Aorto-oesophageal and aortobronchial fistulae following thoracic endovascular aortic repair: a national survey.

            R Chiesa, G Melissano, E.M. Marone … (2010)
            We evaluated the incidence of aorto-oesophageal (AEF) and aortobronchial (ABF) fistulae after thoracic endovascular aortic repair (TEVAR), and investigated their clinical features, determinants, therapeutic options and results. We conducted a voluntary national survey among Italian universities and hospital centres with a thoracic endovascular programme. Thirty-nine centres were contacted, and 17 participated. Of the patients who underwent TEVAR between 1998 and 2008, 19/1113 (1.7%) developed AEF/ABF. Among indications to TEVAR, aortic pseudo-aneurysm was associated with the development of late AEF/ABF (P = 0.009). Further, emergent and complicated procedures resulted in increased risk of AEF/ABF (P = 0.008 and P < 0.001, respectively). Eight patients were treated conservatively, all of whom died within 30 days. Eleven patients underwent AEF/ABF surgical treatment, with a perioperative mortality of 64% (7/11). At a mean follow-up of 17.7 +/- 12.5 months, overall survival was 16% (3/19). The incidence of AEF and ABF following TEVAR is not negligible, and is comparable to that following open repair. This finding warrants an ad hoc long-term follow-up after TEVAR, particularly in patients submitted to emergent and complicated procedures. Both surgical and endovascular treatment of AEF/ABF are associated with high mortality. However, conservative treatment does not appear to be a viable option. Copyright 2009 European Society for Vascular Surgery. Published by Elsevier Ltd. All rights reserved.
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              Aorto-bronchial and aorto-pulmonary fistulation after thoracic endovascular aortic repair: an analysis from the European Registry of Endovascular Aortic Repair Complications

              Martin Czerny, Diana Reser, Holger Eggebrecht … (2015)
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                Author and article information

                Contributors
                Yu-san Chien: cyusan@gmail.com
                Yen-Chun Chao: fayfayfalin@yahoo.com.tw
                Kuo-Sheng Lee: kslee147@ms31.hinet.net
                Kang-Hong Hsu:
                ORCID: http://orcid.org/0000-0003-3191-9813
                erythlet@gmail.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): 31 March 2021
                Publication date PMC-release: 31 March 2021
                Publication date Collection: 2021
                Volume: 16
                Electronic Location Identifier: 62
                Affiliations
                [1 ]Department of Critical Care Mackay Memorial Hospital, CVICU-B, 7F, No. 92, Sec. 2, Zhongshan N. Rd, Taipei City, Taiwan
                [2 ]Department of Pediatric Cardiology Mackay Children’s Hospital, PICU, 11F, No. 92, Sec. 2, Zhongshan N. Rd, Taipei City, Taiwan
                [3 ]GRID grid.413593.9, ISNI 0000 0004 0573 007X, Department of Otorhinolaryngology and Head & Neck Surgery Mackay Memorial Hospital, ; No. 92, Sec. 2, Zhongshan N. Rd, Taipei City, Taiwan
                [4 ]Department of Cardiovascular Surgery Mackay Memorial Hospital, CVICU-B, 7F, No. 92, Sec. 2, Zhongshan N. Rd, Taipei City, Taiwan
                Author information
                http://orcid.org/0000-0003-3191-9813
                Article
                Publisher ID: 1438
                DOI: 10.1186/s13019-021-01438-6
                PMC ID: 8011094
                PubMed ID: 33789718
                SO-VID: 40a5d033-65b7-4b8c-ba55-7675b57ef1a2
                Copyright © © The Author(s) 2021
                License:

                Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. 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 in a credit line to the data.

                History
                Date received : 27 April 2020
                Date accepted : 16 March 2021
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2021

                ScienceOpen disciplines: Surgery
                Keywords: aortotracheal fistula,slide tracheoplasty,dextrocardia,case report
                Data availability:
                ScienceOpen disciplines: Surgery
                Keywords: aortotracheal fistula, slide tracheoplasty, dextrocardia, case report

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