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Adult Benign, Non-Iatrogenic Bronchoesophageal Fistulae: Systematic Review and Descriptive Analysis of Individual Patient Data

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Abstract

Background

Adult, benign, non-iatrogenic bronchoesophageal fistula (BEF) is a rare condition, which is occasionally described in single case reports. Therefore, little is known about its possible causes, presentation and management.

Methods

A systematic search of the literature in MEDLINE, PubMed Central and EMBASE databases between 1990 and 2020 was carried out to identify all cases of BEF. The initial database search identified 19,452 articles, of which 183 (251 individual patient cases) were included in the final analysis.

Results

Main causes of BEF were congenital malformations (97/251, 38.7%) and infections (82/251, 32.7%), while 33/251 (13.1%) fistulae were regarded as idiopathic and 39/251 (15.5%) attributed to other causes. Esophagograpy was the most sensitive method of diagnosis (97.4%) compared with esophagoscopy (78.9%), computed tomography (49.6%) and bronchoscopy (46.0%). Definitive treatment was surgical for 176 patients (70%), endoscopic for 25 (10%) and medical for 37 (14.7%). Compared with congenital BEFs, infective BEFs had shorter median symptom duration and were distributed more proximally over the bronchial tree. Definitive treatment was almost only surgical for congenital BEFs, while infective BEFs were treated also endoscopically (12%) and by medical therapy (38%). Morbidity, treatment failure and recurrence rates were higher for infective BEFs.

Conclusions

BEFs are rare. Symptoms are non-specific and a high index of suspicion is necessary for diagnosis. Patients with infective BEF tend to have a more severe clinical picture than those with congenital BEF. Surgery is the main treatment for patients affected by congenital BEF, while infective BEFs may heal conservatively.

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References

  1. Balazs A, Galambos Z, Kupcsulik PK (2009) Characteristics of esophagorespiratory fistulas resulting from esophageal cancers: a single-center study on 243 cases in a 20-year period. World J Surg 33:994–1001

    Article  Google Scholar 

  2. Yasuda T, Sugimura K, Yamasaki M et al (2012) Ten cases of gastro-tracheobronchial fistula: a serious complication after esophagectomy and reconstruction using posterior mediastinal gastric tube. Dis Esophagus 25:687–693

    Article  CAS  Google Scholar 

  3. Faro RS, Goodwin CD, Jr Organ CH et al (1979) Tracheal agenesis. Ann Thorac Surg 28:295–299

    Article  CAS  Google Scholar 

  4. Demircan M, Aksoy T, Ceran C et al (2008) Tracheal agenesis and esophageal atresia with proximal and distal bronchoesophageal fistulas. J Pediatr Surg 43:e1–e3

    Article  Google Scholar 

  5. Risher WH, Arensman RM, Ochsner JL (1990) Congenital bronchoesophageal fistula. Ann Thorac Surg 49:500–505

    Article  CAS  Google Scholar 

  6. Lado Lado FL, Golpe Gómez A, Ortíz C, de Barrón A et al (2002) Bronchoesophageal fistulae secondary to tuberculosis. Respiration 69:362–365

    Article  CAS  Google Scholar 

  7. Moher D, Liberati A, Tetzlaff J, Altman DG, PRISMA Group (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. BMJ 339:b2535

    Article  Google Scholar 

  8. Braimbridge MV, Keith HI (1965) Oesophago-bronchial fistula in the adult. Thorax 20:226–233

    Article  CAS  Google Scholar 

  9. Ballehaninna UK, Shaw JP, Brichkov I (2012) Traction esophageal diverticulum: a rare cause of gastro-intestinal bleeding. Springerplus 1:50

    Article  Google Scholar 

  10. Riquet M, Pricopi C, Le Pimpec BF (2013) Fistule œso-bronchique séquelle d’une atteinte œsophagienne tuberculeuse [Esophagobronchial fistula as an end complication of esophageal tuberculosis]. Rev Pneumol Clin 69:305–306

    Article  CAS  Google Scholar 

  11. Morse CR (2009) Repair of bronchial esophageal fistula using alloderm. Ann Thorac Surg 88:1018–1019

    Article  Google Scholar 

  12. Ho IK, Guarino DP, Pertsovskiy Y et al (2002) Infliximab treatment of an esophagobronchial fistula in a patient with extensive Crohn’s disease of the esophagus. J Clin Gastroenterol 34:488–489

    Article  Google Scholar 

  13. Klein M, Neubert U, Koop H (2000) Esophagobronchial fistula combined with a peptic esophageal stenosis. Z Gastroenterol 38:841–844

    Article  CAS  Google Scholar 

  14. Kim JH, Park KH, Sung SW (1995) Congenital bronchoesophageal fistulas in adult patients. Ann Thorac Surg 60:151–155

    Article  CAS  Google Scholar 

  15. Ahn JY, Jung HY, Choi JY et al (2010) Benign bronchoesophageal fistula in adults: endoscopic closure as primary treatment. Gut Liver 4:508–513

    Article  Google Scholar 

  16. Kim HK, Choi YS, Kim K et al (2007) Long-term results of surgical treatment in benign bronchoesophageal fistula. J Thorac Cardiovasc Surg 134:411–414

    Article  Google Scholar 

  17. Zhang BS, Zhou NK, Yu CH (2011) Congenital bronchoesophageal fistula in adults. World J Gastroenterol 17:1358–1361

    Article  Google Scholar 

  18. Mangi AA, Gaissert HA, Wright CD et al (2002) Benign broncho-esophageal fistula in the adult. Ann Thorac Surg 73:911–915

    Article  Google Scholar 

  19. Brunner A (1961) Esophagobronchial fistulae. Munch Med Wochenschr 103:2181–2184

    CAS  PubMed  Google Scholar 

  20. Vachtenheim J Jr, Lischke R (2019) Esophageal bypass surgery as a definitive repair of recurrent acquired benign bronchoesophageal fistula. J Cardiothorac Surg 14:73

    Article  Google Scholar 

  21. Black AD, Bizos D, Perovic O (2001) Broncho-oesophageal fistula due to idiopathic oesophageal ulceration in a patient with AIDS. S Afr Med J 91:116–117

    CAS  PubMed  Google Scholar 

  22. Kinoshita S, Nishizawa T, Hisamatsu T et al (2017) Polyglycolic acid sheet for closure of esophagobronchial fistula in a patient with Behçet’s disease. Gastrointest Endosc 85:1094–1096

    Article  Google Scholar 

  23. Alkhuja S, Miller A (1998) Tuberculous bronchoesophageal fistulae in patients infected with the human immunodeficiency virus: a case report and review. Heart Lung 27:143–145

    Article  CAS  Google Scholar 

  24. Hu XF, Chen C, Duan L et al (2011) Aspiration via congenital broncho-esophageal fistula after lobectomy. Respir Care 56:1195–1197

    Article  Google Scholar 

  25. Parry GW, Juma A, Dussek JE (1993) Broncho-oesophageal fistula treated effectively without surgical resection. Thorax 48:189–190

    Article  CAS  Google Scholar 

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Authors and Affiliations

  1. Thoracic Surgery Division, Department of Cardiac, Thoracic, Vascular Sciences and Public Health, Padova University Hospital, Via Giustiniani, 2, 35128, Padua, Italy

    Marco Mammana, Alessandro Pangoni, Chiara Catelli, Giuseppe Natale, Andrea Dell’Amore & Federico Rea

  2. Unit of Biostatistics, Epidemiology and Public Health, Department of Cardiac, Thoracic, Vascular Sciences and Public Health, Padua University Hospital, Padua, Italy

    Giulia Lorenzoni & Dario Gregori

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  1. Marco Mammana
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  2. Alessandro Pangoni
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  3. Giulia Lorenzoni
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  4. Chiara Catelli
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  5. Giuseppe Natale
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  7. Andrea Dell’Amore
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  8. Federico Rea
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Correspondence to Marco Mammana.

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Mammana, M., Pangoni, A., Lorenzoni, G. et al. Adult Benign, Non-Iatrogenic Bronchoesophageal Fistulae: Systematic Review and Descriptive Analysis of Individual Patient Data. World J Surg 45, 3449–3457 (2021). https://doi.org/10.1007/s00268-021-06266-2

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  • DOI: https://doi.org/10.1007/s00268-021-06266-2

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