10.14639/0392-100X-N2517
text/xml
1827-675X
2517
en
Francisco Hernández Oliveros
http://creativecommons.org/licenses/by-nc-nd/4.0
ACTA Otorhinolaryngologica Italica
airway reconstruction
Type III laryngo-tracheo-oesophageal cleft repair without tracheostomy in neonate with long-gap oesophageal atresia
Text.Serial.Journal
https://www.actaitalica.it/
43
Letters to the Editor-in-Chief
2023-07-24
2023-02-12
2023-07-25
2023-07-25
291-293
1
Francisco Hernández Oliveros
2023/07/24
10.14639/0392-100X-N2517
291
Department of Pediatric Surgery, La Paz Children’s University Hospital, Madrid, Spain; Pediatric Airway Unit, La Paz Children’s University Hospital, Madrid, Spain
1827-675X
airway reconstruction
en
293
Type III laryngo-tracheo-oesophageal cleft repair without tracheostomy in neonate with long-gap oesophageal atresia
https://www.actaitalica.it/article/download/2517/1020
43
ACTA OTORHINOLARYNGOLOGICA ITALICA 2023;43:291-293; doi: 10.14639/0392-100X-N2517




Letter to the Editor


Type III laryngo-tracheo-oesophageal cleft repair
without tracheostomy in neonate with long-gap
oesophageal atresia
Riparazione di una fistola laringo-tracheo-esofagea di tipo III senza tracheostomia
in neonato con atresia esofagea estesa
Carlos Delgado-Miguel1, Carlos De la Torre2, Luis Castro2,3, Francisco Hernández Oliveros1,2
1
 Department of Pediatric Surgery, La Paz Children’s University Hospital, Madrid, Spain; 2 Pediatric Airway Unit, La Paz Children’s
University Hospital, Madrid, Spain; 3 Department of Pediatric Anaesthesiology and Resuscitation, La Paz Children’s University
Hospital, Madrid, Spain



KEY WORDS: laryngo-tracheo-esophageal cleft, long-gap esophageal atresia, airway
reconstruction
PAROLE CHIAVE: fistola laringo-tracheo-esofagea, atresia esofagea estesa, ricostruzione
delle vie aeree


Dear Editor,
Laryngo-tracheo-oesophageal cleft (LTEC) is a rare congenital anomaly that
involves a communication between the upper respiratory and digestive tracts
as a consequence of an alteration in tracheo-oesophageal septum develop-
ment 1. LTEC is divided into 4 types depending on its extension according
to the Benjamin and Inglis classification: type I (supraglottic interarytenoid
involvement), type II (partial involvement of the cricoid lamina), type III (total
involvement of the cricoid lamina and cervical trachea) and type IV (intratho-
racic trachea involvement) 2. This congenital malformation is frequently asso-            Received: February 12, 2023
                                                                                          Accepted: March 17, 2023
ciated with other anomalies in up to 60% of cases, of which tracheoesophageal
fistula, oesophageal atresia, and tracheomalacia stand out 1. Mortality has been          Correspondence
attributed directly to associated comorbidities such as poor feeding and aspira-          Carlos Delgado-Miguel
tion pneumonia, regardless of surgical treatment 3. In terms of post-operative            Department of Pediatric Surgery, La Paz Children’s
                                                                                          Hospital, Paseo de la Castellana 261, Madrid, Spain
complications, fistula re-formation, recurrent aspiration and tracheomalacia              E-mail: carlosdelgado84@hotmail.com
are common, and require close follow-up for early diagnosis and treatment 4.
We describe a male born at 36 + 5 weeks of gestational age (2,350 grams),                   How to cite this article: Delgado-Miguel C, De la
delivered by emergency caesarean section due to loss of foetal well-being,                  Torre C, Castro L, et al. Type III laryngo-tracheo-
with a prenatal diagnosis of severe polyhydramnios. After patient stabilisa-                oesophageal cleft repair without tracheostomy in
                                                                                            neonate with long-gap oesophageal atresia. Acta
tion, fibrescopic airway examination revealed a LTEC with cervical tracheal                 Otorhinolaryngol Ital 2023;43:291-293. https://doi.
involvement up to 2 cm from the carina (type III) and an atresic oesophageal                org/10.14639/0392-100X-N2517
pouch 2 cm in length, compatible with long-gap oesophageal atresia. Distal
                                                                                          © Società Italiana di Otorinolaringoiatria
gas in abdominal X-ray confirmed the diagnosis of tracheo-oesophageal fis-                e Chirurgia Cervico-Facciale
tula (TEF) type C according to the Gross classification (type A, isolated oe-
sophageal atresia; type B, proximal fistula with distal atresia; type C, proximal                        OPEN ACCESS
atresia with distal fistula; type D, double fistula with intervening atresia and          This is an open access article distributed in accordance with
                                                                                          the CC-BY-NC-ND (Creative Commons Attribution-Non-
type E, isolated fistula) 5. Physical exam showed anal atresia with bilateral             Commercial-NoDerivatives 4.0 International) license. The
thumb amputation. In addition, abdominal ultrasound found left renal agen-                article can be used by giving appropriate credit and mentio-
                                                                                          ning the license, but only for non-commercial purposes and
esis, all these findings compatible with VACTERL polymalformative syn-                    only in the original version. For further information: https://
drome (vertebral defects, anal atresia, cardiac defects, TEF, renal anomalies,            creativecommons.org/licenses/by-nc-nd/4.0/deed.en




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C. Delgado-Miguel et al.




                                                                            Different approaches have been described for the repair of
                                                                            type III-IV LTEC. Anterior cervical approach allows re-
                                                                            construction from an extended cervicotomy. In this case,
                                                                            the most difficult step was the separation of the posterior
                                                                            tracheal and anterior oesophageal walls. After complete
                                                                            reconstruction of upper oesophageal pouch, a left cervi-
                                                                            cal oesophagostomy was performed, in order to allow
                                                                            adequate saliva outflow after swallowing, reducing the
                                                                            risk of aspiration pneumonia and refistulisation. In fact,
                                                                            one of the major problems after LTEC repair is the occur-
                                                                            rence of fistulas between the trachea and the oesophagus.
                                                                            Therefore, several techniques have been described to try
                                                                            to reduce this risk, such as the interposition of sterno-
                                                                            cleidomastoid muscle flap and three layers of closure 6.
                                                                            Our patient also had an associated type III TEF, which
                                                                            had been previously ligated. We decided to perform a
                                                                            tibial periosteal graft interposition between the oesopha-
                                                                            gus and trachea to prevent possible refistulisation. This
                                                                            avoided the use of other cervical structures such as the
                                                                            sternocleidomastoid muscle, which would have made oe-
Figure 1. Bronchoscopic features before LTEC repair (A-B) and 2 weeks af-   sophagostomy performance more difficult.
ter surgical repair (C-D).                                                  Tracheotomy is widely used for type III and IV LTEC for
                                                                            protection of the lung against aspiration during enteral
                                                                            feeding. In our case, we chose to avoid performing initial
and limb abnormalities). The TEF was ligated on the first                   tracheostomy. Therefore, once the anterior tracheal dissec-
day of life through an abdominal approach (supraumbili-                     tion was completed, the anterior tracheal wall was opened
cal midline laparotomy), and a 10 Fr Pezzer gastrostomy                     longitudinally and a ventilatory endotracheal tube was
tube was placed following Stamm’s technique. In the same                    placed in the healthy trachea distal to the LTEC. Airway
intervention, a sigmoidostomy was performed, with meco-                     management of neonates with tracheo-oesophageal anoma-
nium passing through it 24 hours after surgery.                             lies presents a challenge for neonatologists and anaesthesi-
The LTEC was repaired at two months of age, when a weight                   ologists. Because neonates with this condition are usually
of 4500 grams had been reached. An anterior cervical approach               at risk of life-threatening respiratory problems, difficult
was performed, with anterior tracheal opening, followed by                  intubation must be generally expected 3. In our patient,
reconstruction of the upper oesophageal pouch by separation                 the presence of tracheomalacia posed an added difficulty,
from surrounding structures to achieve closure of the anterior              which is being managed by biodegradable tracheal stents.
oesophageal wall by continuous suture in one plane. After                   In most cases, tracheomalacia resolves spontaneously at
tubular reconstruction, a left cervical oesophagostomy was                  around 18-24 months, after completion of tracheal cartilage
performed. Subsequently, a 30 x 10 mm anterior tibial peri-                 maturation. For this reason, repair of the oesophagus will
osteal graft was interposed between the oesophageal wall and                be performed once definitive airway stability is achieved.
the posterior tracheal wall. Finally, the posterior and anterior            The type of oesophageal reconstruction with stomach or
tracheal walls were sutured, achieving complete repair of the               colon (gastric pull-up or oesophagocoloplasty respectively)
defect, with extubation on the 7th post-operative day without               will be also influenced by the associated anorectal malfor-
complications (Fig. 1). Bronchoscopic exams were periodical-                mation (ARM). In the next operation, ARM correction will
ly performed in which the tracheoplasty remained satisfactori-              be performed initially and, subsequently, the oesophageal
ly repaired, although a moderate degree of tracheomalacia and               reconstruction option will be decided upon depending on
partial collapse of the airway were observed. For this reason,              the availability of the colon after ARM repair.
a biodegradable tracheal stent (30 x 6 mm) was placed, which                To sum up, repair of types III-IV LTEC should be con-
was replaced on 2 subsequent occasions, every 3 months un-                  sidered a surgical challenge that requires complex airway
til the present day. At present, the patient is 12 months old               management, and should therefore be performed in expe-
(10.5 kg), and is growing properly at home, with follow-up                  rienced centres with paediatric aerodigestive surgery pro-
bronchoscopies every 6-8 weeks.                                             grammes.



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Acknowledgements                                                        Child’s parents signed a written informed consent form,
We would like to thank all the hospital staff who have con-             which included the publication of the images of this case.
tributed to the care of the patient from birth to the present
day.                                                                    References
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Conflict of interest statement                                              clinical outcome of laryngotracheal clefts types III and IV: systematic
The authors declare no conflict of interest.                                analysis of new cases. Pediatr Pulmonol 2021;56:138-144. https://doi.
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Funding                                                                 2
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This research did not receive any specific grant from fund-                 ps://doi.org/10.1177/000348948909800603
ing agencies in the public, commercial, or not-for-profit               3
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Author contributions                                                    4
                                                                            Kawaguchi AL, Donahoe PK, Ryan DP. Management and long-term
                                                                            follow-up of patients with types III and IV laryngotracheoesophage-
CDM: designed and conceptualised the study and wrote the
                                                                            al clefts. J Pediatr Surg 2005;40:158-164. https://doi.org/10.1016/j.
draft manuscript; CDT, LC: performed data adquisition and                   jpedsurg.2004.09.041
data analysis; CDT, FHO: critically revised the manuscript.             5
                                                                            Yang S, Yang R, Ma X, et al. Detail correction for Gross classification
All authors approved the final version of the manuscript.                   of esophageal atresia based on 434 cases in China. Chin Med J (Engl)
                                                                            2021;135:485-487. https://doi.org/10.1097/CM9.0000000000001673
Ethical consideration
                                                                        6
                                                                            Geller K, Kim Y, Koempel J, et al. Surgical management of type
                                                                            III and IV laryngotracheoesophageal clefts: the three-layered ap-
This study obtained the approval of the institutional eth-                  proach. Int J Pediatr Otorhinolaryngol 2010;74:652-657. https://doi.
ics committee and the hospital review board (PI-502788).                    org/10.1016/j.ijporl.2010.03.013




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