Skip to main content

Advertisement

SpringerLink
Log in

Clinical features of a tracheoesophageal fistula associated with complete tracheal rings

  • Original Article
  • Published:
Pediatric Surgery International Aims and scope Submit manuscript

Abstract

Purpose

A tracheoesophageal fistula (TEF) associated with complete tracheal rings (CTR) is extremely rare. This study aimed to elucidate the clinical features of this combination.

Methods

Records of 39 patients diagnosed with TEF with or without CTR between January 2013 and February 2023 were retrospectively reviewed. Data collected included location of the TEF and CTR, esophageal gap, surgery of TEF/esophageal atresia (EA), and symptoms of CTR.

Results

Seven patients had CTR, while 32 patients did not have CTR. TEF was frequently located higher than the T4 vertebra, and the median esophageal gap was shorter in patients with CTR than in those without (5/7 [71.4%] vs. 6/32 [18.8%], P = 0.012; 0 (range: 0–15) mm vs. 13.3 (range: 0–40) mm, P = 0.017, respectively). TEF was located just above the beginning of the CTR in all patients. All patients with CTR underwent primary esophageal anastomosis. Additionally, two patients were diagnosed with CTR before TEF/EA repair. A tracheal tube was prevented from contacting the CTR during TEF/EA repair, and respiratory insufficiency did not occur.

Conclusions

Our results may help pediatric surgeons in the management of TEF with CTR.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1

Similar content being viewed by others

Data availability

Not applicable.

References

  1. Shaw-Smith C (2006) Oesophageal atresia, tracheo-oesophageal fistula, and the VACTERL association: review of genetics and epidemiology. J Med Genet 43:545–554. https://doi.org/10.1136/jmg.2005.038158

    Article  CAS  PubMed  Google Scholar 

  2. Rothenberg S (2014) Esophageal atresia and tracheoesophageal fistula malformations. In: HolcombMurphy GWJP, Ostlie DJ (eds) Ashcraft’s pediatric surgery. Elsevier Inc, London, pp 365–384

    Google Scholar 

  3. Hofferberth SC, Watters K, Rahbar R, Fynn-Thompson F (2015) Management of congenital tracheal stenosis. Pediatrics 136:e660-669. https://doi.org/10.1542/peds.2014-3931

    Article  PubMed  Google Scholar 

  4. Herrera P, Caldarone C, Forte V, Campisi P, Holtby H, Chait P, Chiu P, Cox P, Yoo SJ, Manson D, Kim PC (2007) The current state of congenital tracheal stenosis. Pediatr Surg Int 23:1033–1044. https://doi.org/10.1007/s00383-007-1945-3

    Article  CAS  PubMed  Google Scholar 

  5. Chiu PP, Kim PC (2006) Prognostic factors in the surgical treatment of congenital tracheal stenosis: a multicenter analysis of the literature. J Pediatr Surg 41:221–225. https://doi.org/10.1016/j.jpedsurg.2005.10.043

    Article  PubMed  Google Scholar 

  6. Borland LM, Reilly JS, Smith SD (1987) Anesthetic management of tracheal esophageal fistula with distal tracheal stenosis. Anesthesiology 67:132–133. https://doi.org/10.1097/00000542-198707000-00030

    Article  CAS  PubMed  Google Scholar 

  7. Usui N, Kamata S, Ishikawa S, Sawai T, Okuyama H, Imura K, Okada A (1996) Anomalies of the tracheobronchial tree in patients with esophageal atresia. J Pediatr Surg 31:258–262. https://doi.org/10.1016/s0022-3468(96)90010-x

    Article  CAS  PubMed  Google Scholar 

  8. Lam WW, Tam PK, Chan FL, Chan KL, Cheng W (2000) Esophageal atresia and tracheal stenosis: use of three-dimensional CT and virtual bronchoscopy in neonates, infants, and children. Am J Roentgenol 174:1009–1012. https://doi.org/10.2214/ajr.174.4.1741009

    Article  CAS  Google Scholar 

  9. Rutter MJ, Cotton RT, Azizkhan RG, Manning PB (2003) Slide tracheoplasty for the management of complete tracheal rings. J Pediatr Surg 38:928–934. https://doi.org/10.1016/s0022-3468(03)00126-x

    Article  PubMed  Google Scholar 

  10. Seguier-Lipszyc E, Dauger S, Malbezin S, Aigrain Y, de Lagausie P (2005) Reimplantation of oesophageal bronchus following a type III oesophageal atresia repair. Pediatr Surg Int 21:649–651. https://doi.org/10.1007/s00383-004-1359-4

    Article  CAS  PubMed  Google Scholar 

  11. Tsugawa J, Tsugawa C, Satoh S, Nishijima E, Muraji T, Takamizawa S, Kanegawa K, Akasaka Y (2005) Communicating bronchopulmonary foregut malformation: particular emphasis on concomitant congenital tracheobronchial stenosis. Pediatr Surg Int 21:932–935. https://doi.org/10.1007/s00383-005-1520-8

    Article  PubMed  Google Scholar 

  12. Le Bret E, Roubertie F, Roger G, Sigal-Cinqualbre A, Coblence M, Belli E, Gharbi N, Roussin R, Garabédian EN, Serraf A (2009) Slide tracheoplasty in infant with congenital tracheal stenosis and tracheomalacia after esophageal atresia with tracheoesophageal fistula repair. J Pediatr Surg 44:e15-17. https://doi.org/10.1016/j.jpedsurg.2009.04.004

    Article  PubMed  Google Scholar 

  13. Antón-Pacheco JL, Kalicinski P, Kansy A, Maruszewski P (2012) Slide tracheoplasty in an infant with congenital tracheal stenosis and oesophageal atresia with tracheoesophageal fistula. Eur J Cardiothorac Surg 42:892–893. https://doi.org/10.1093/ejcts/ezs354

    Article  PubMed  Google Scholar 

  14. Wu ET, Wang CC, Lin MT, Huang PM, Chen SJ, Huang CH, Hwang HK, Chen MR, Huang SC (2013) Concomitant slide tracheoplasty and cardiac operation for congenital tracheal stenosis associated with VACTERL. Ann Thorac Surg 96:1492–1495. https://doi.org/10.1016/j.athoracsur.2013.04.122

    Article  PubMed  Google Scholar 

  15. Stock C, Nathan M, Murray R, Rahbar R, Fynn-Thompson F (2015) Modified end-to-end anastomosis for the treatment of congenital tracheal stenosis with a bridging bronchus. Ann Thorac Surg 99:346–348. https://doi.org/10.1016/j.athoracsur.2014.06.084

    Article  PubMed  Google Scholar 

  16. Siddiqui A, Wolter NE, Matava C (2020) Use of an aortic cannula for tracheal intubation in a patient with severe tracheal stenosis and tracheoesophageal fistula: a case report. Cureus 12:e9456. https://doi.org/10.7759/cureus.9456

    Article  PubMed  PubMed Central  Google Scholar 

  17. Morita K, Kawahara I, Yashita H, Hatakeyama T (2022) Right esophageal lung with esophageal atresia and left bronchial stenosis. Pediatr Int 64:e14982. https://doi.org/10.1111/ped.14982

    Article  PubMed  Google Scholar 

  18. Wolter NE, Kennedy AA, Rutter MJ, Matava C, Honjo O, Chiu PL, Propst EJ (2020) Diagnosis and management of complete tracheal rings with concurrent tracheoesophageal fistula. Int J Pediatr Otorhinolaryngol 133:109971. https://doi.org/10.1016/j.ijporl.2020.109971

    Article  PubMed  Google Scholar 

  19. Holzki J (1992) Bronchoscopic findings and treatment in congenital tracheoesophageal fistula. Paediatr Anaesth 2:297–303. https://doi.org/10.1111/j.1460-9592.1992.tb00220.x

    Article  Google Scholar 

  20. Shimojima N, Shimotakahara A, Tomita H, Harumatsu T, Harada A, Maeda Y, Ito Y, Miyaguni K, Tsukizaki A, Abe K, Hashimoto M, Ishikawa M, Honda M, Kuroda T, Hirobe S (2022) Outcomes of slide tracheoplasty for congenital tracheal stenosis in 80 children: a 22-year single-center experience. J Pediatr Surg 57:1205–1209. https://doi.org/10.1016/j.jpedsurg.2022.02.033

    Article  PubMed  Google Scholar 

  21. Taghavi K, Stringer MD (2018) Preoperative laryngotracheobronchoscopy in infants with esophageal atresia: why is it not routine? Pediatr Surg Int 34:3–7. https://doi.org/10.1007/s00383-017-4194-0

    Article  PubMed  Google Scholar 

  22. Sharma N, Srinivas M (2014) Laryngotracheobronchoscopy prior to esophageal atresia and tracheoesophageal fistula repair-its use and importance. J Pediatr Surg 49:367–369. https://doi.org/10.1016/j.jpedsurg.2013.09.009

    Article  PubMed  Google Scholar 

  23. Antón-Pacheco JL, Cano I, Comas J, Galletti L, Polo L, García A, López M, Cabezalí D (2006) Management of congenital tracheal stenosis in infancy. Eur J Cardiothorac Surg 29:991–996. https://doi.org/10.1016/j.ejcts.2005.12.061

    Article  PubMed  Google Scholar 

  24. Tsang V, Murday A, Gillbe C, Goldstraw P (1989) Slide tracheoplasty for congenital funnel-shaped tracheal stenosis. Ann Thorac Surg 48:632–635. https://doi.org/10.1016/0003-4975(89)90777-7

    Article  CAS  PubMed  Google Scholar 

  25. Grillo HC (1994) Slide tracheoplasty for long-segment congenital tracheal stenosis. Ann Thorac Surg 58:613–619. https://doi.org/10.1016/0003-4975(94)90714-5

    Article  CAS  PubMed  Google Scholar 

  26. Morita K, Maeda K, Yabe K, Oshima Y (2017) Management of congenital tracheal stenosis in the neonatal period. Pediatr Surg Int 33:1059–1063. https://doi.org/10.1007/s00383-017-4137-9

    Article  PubMed  Google Scholar 

  27. Garge S, Rao KL, Bawa M (2013) The role of preoperative CT scan in patients with tracheoesophageal fistula: a review. J Pediatr Surg 48:1966–1971. https://doi.org/10.1016/j.jpedsurg.2013.06.010

    Article  PubMed  Google Scholar 

  28. Garge S, Rao KL, Bawa M (2013) Role of preoperative 3D CT reconstruction for evaluation of patients with esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 48:1966–1971. https://doi.org/10.1016/j.jpedsurg.2013.06.010

    Article  PubMed  Google Scholar 

Download references

Funding

This research received no specific grant from any funding agency belonging to public, commercial, or not-for-profit sectors.

Author information

Authors and Affiliations

  1. Department of Pediatric Surgery, Kobe Children’s Hospital, 1-6-7 Minatojimaminamimachi, Chuo-ku, Kobe, 650-0047, Japan

    Keiichi Morita & Tadashi Hatakeyama

Authors
  1. Keiichi Morita
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Tadashi Hatakeyama
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

Conceptualization, methodology, formal analysis, investigation, and writing: KM; supervision: TH.

Corresponding author

Correspondence to Keiichi Morita.

Ethics declarations

Conflict of interest

The authors declare that they have no conflict of interest.

Ethical approval

This study has been approved by the institutional ethics committee (approval number: R4-113) and performed in line with the principles of the Declaration of Helsinki.

Consent to participate and consent to publish

This study was open to the public and guaranteed a refusal to cooperate; the data were anonymous, and the need for a separate informed consent was waived.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Morita, K., Hatakeyama, T. Clinical features of a tracheoesophageal fistula associated with complete tracheal rings. Pediatr Surg Int 39, 218 (2023). https://doi.org/10.1007/s00383-023-05503-0

Download citation

  • Accepted:

  • Published:

  • DOI: https://doi.org/10.1007/s00383-023-05503-0

Keywords

Search

Navigation