Isolated Congenital Tracheal Stenosis in a 2-Year-Old Female: A Case Report

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Case Report

Isolated Congenital Tracheal Stenosis in a 2-Year-Old Female: A Case Report

https://doi.org/10.21203/rs.3.rs-4297011/v1

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Background: Congenital tracheal stenosis (CTS) is a rare but potentially life-threatening pediatric condition characterized by narrowing of the trachea. This narrowing leads to significant breathing difficulties, often accompanied by recurrent respiratory infections. CTS frequently coexists with other congenital malformations, particularly those affecting the heart and pulmonary artery. However, isolated cases where CTS occurs without accompanying anomalies are exceptionally rare and pose unique diagnostic and treatment challenges. This report describes the case of a 2-year-old female who was initially misdiagnosed and treated for laryngomalacia and asthma. However, her symptoms were not alleviated due tounderlying, undiagnosed isolated CTS.

Case Presentation: This case involved a 2-year-old female with a history of breathing difficulties, productive cough, and recurrent pneumonia who was treated for laryngomalacia and asthma and who presented with persistent respiratory distress and dysmorphic features. Imaging, including laryngoscopy and chest CT scans, revealed distal tracheal narrowing. The diagnosis was tracheobronchitis secondary to distal congenital tracheal stenosis with no other associated cardiac or respiratory congenital malformations. Surgical intervention entailed resection of the stenotic segment and anastomosis, followed by intensive postoperative care in the ICU and a structured plan for bronchoscopy dilation procedures. Patient recovery requires successful surgical correction and emphasizes the critical role of multidisciplinary care.

Conclusion: This report underscores the crucial significance of promptly identifying and accurately managing isolated congenital stenosis in children. This highlights the risk of misdiagnosis due to nonspecific respiratory symptoms and showcases the effectiveness of a multidisciplinary treatment approach, resulting in a successful outcome.

Congenital Tracheal Stenosis

Pediatric Respiratory Distress

Isolated Congenital Tracheal Stenosis

Diagnostic Challenges

Respiratory Infections

Imaging in Airway Diseases

Congenital tracheal stenosis (CTS), although a rare condition, poses a significant threat to life, with an estimated incidence of CTS of approximately 1 for every 64,500 live births (1). This condition is not isolated in its complexity; more than half of those affected also suffer from concurrent congenital anomalies, predominantly cardiovascular in nature, such as left pulmonary artery sling, patent ductus arteriosus, ventricular septal defect, and intricate congenital heart diseases (1–5). Moreover, the association of CTS with pulmonary agenesis or hypoplasia adds layers of complexity to its management (5,6). However, in approximately 10% to 30% of cases, CTS occurs in isolation (7).

The defining characteristic CTS is the constriction of the trachea, precipitating a spectrum of respiratory manifestations and distresses (8). These include inspiratory and expiratory stridor, recurrent infections, and potentially life-threatening complications, if not adequately addressed. A significant proportion of patients present with various respiratory symptoms of differing severity, with a substantial number necessitating urgent hospitalization following episodes of collapse at home, during which parental resuscitation is often imperative (9–12). The management of CTS can be exceptionally challenging, with difficulties in intubation and ventilation, at times necessitating the use of extracorporeal membrane oxygenation (ECMO) for respiratory support (13–15). Hence, the early identification and meticulous management of CTS are critical for preventing long-term respiratory complications and improving patient outcomes.

The gold standard for diagnosing CTS is rigid bronchoscopy under general anesthesia. This procedure not only facilitates visualization of tracheal anatomy and precise identification of the location of stenosis but also evaluates the dynamic behavior and volume of the unaffected airway, thereby playing a crucial role in treatment planning (16,17). During the bronchoscopy procedure, meticulous attention is imperative to prevent the induction of minor mucosal edema at the stenotic segment, which could result in severe airway obstruction (1,16). The initial diagnosis typically begins with contrast-enhanced computed tomography (CT) imaging, which offers detailed insights essential for ruling out external compression of the trachea by masses or vascular anomalies and is confirmed by bronchoscopy (10,18). CT scans, especially when combined with multiplanar reconstruction (MPR), provide meticulous anatomical delineation of the airway. Concurrently, magnetic resonance imaging (MRI) complements these findings by enabling a comprehensive evaluation of vascular structures and their spatial relationships with the trachea (11).

The management of CTS typically involves a collaborative, multidisciplinary approach in which the choice of treatment is tailored to the patient's clinical status and the specific anatomical features of the stenosis (10). Surgical treatment of CTS depends on the degree of stenosis relative to the total length of the trachea. Stenosis involving less than 30% of the trachea is classified as a short segment, while stenosis exceeding 30% is classified as a long segment (19–21).

For short segments, the primary surgical intervention typically involves tracheal resection with primary anastomosis. This procedure aims to remove the stenotic section and reconnect the healthy ends of the trachea to re-establish a clear airway. In cases of long segment stenosis, slide tracheoplasty is generally considered the treatment of choice. This technique involves making longitudinal cuts in the trachea and sliding the segments over each other to widen the narrow area (22–26).

In less severe cases or as a preliminary step before definitive surgery, nonsurgical interventions, such as balloon dilatation or stent placement, may be appropriate (27,28). The selection of the most suitable therapeutic approach is informed by the patient's clinical presentation, imaging findings, and the expertise of the multidisciplinary team managing the patient's care (29).

This report highlights the case of a 2-year-old female who was initially misdiagnosed and treated for laryngomalacia and asthma. Her symptoms were not relieved due to undiagnosed isolated CTS. This study aimed to improve patient outcomes by emphasizing the importance of considering CTS in pediatric patients with persistent and recurrent respiratory symptoms.

A 2-year-old female patient presented with a history of difficulty breathing associated with noisy breathing and a productive cough since birth. The child was initially managed for laryngomalacia without any significant improvement. She had also been treated previously for pneumonia and asthma on multiple occasions associated with recurrent cyanotic episodes and respiratory distress, although she was reported to be afebrile. There was no history of easy fatiguability or failure to thrive. The mother reported an unremarkable pregnancy and delivered at term, weighing 4.2 kg, via cesarean section due to one previous scar. The patient had met all developmental milestones on time and had received all vaccinations recommended for her age.

Physical Examination upon Arrival:

General Appearance: Alert, with a normal tone and posture, appeared sick and in respiratory distress, not cyanosed, SpO2: 86% on room air. The patients had a weight-for-age z score of +1.5. Head and Neck: Oral examination revealed Grade 3 uninflamed tonsils, with no cervical lymphadenopathy noted. Respiratory System: On inspection, Grade 3 finger clubbing was noted, and symmetrical chest movements with slightly increased AP diameter and tachypnea with increased work of breathing were observed. Biphasic stridor and worsening on expiration were also noted, and there were good bilateral air entry bronchial breath sounds with a prolonged expiratory phase. Low-pitched polyphonic wheezes and widespread coarse crepitations were noted. Dysmorphic features included a depressed nasal bridge, epicanthal folds, low-set ears, polydactyly, and syndactyly.Normal cognitive function and normal cardiovascular parameters

Lung function tests revealed normal lung function.Blood analysis revealed iron deficiency anemia, and chromosomal analysis revealed no chromosomal anomalies.

Normal Echocardiogram

Imaging:Laryngoscopy revealed normal findings. An IV contrast chest CT scanrevealed distal tracheal narrowing before the bifurcation, measuring 7.4 mm in length and 1.4 mm in diameter, and no vascular rings or slings were identified.

Figures 1 and 2 display pre-operative imaging that illustrates narrowing of the distal trachea located proximal to the bifurcation. The constricted segment measures 7.4 mm in length and 1.4 mm in width.

Diagnosis: The patient was diagnosed with tracheobronchitis secondary to distal CTS.

Management: Multidisciplinary management was undertaken to address the complex nature of the patient's condition, involving specialists from various medical fields, including pediatric pulmonologists and pediatric cardiac anesthetists, to ensure comprehensive care and optimal treatment outcomes.

Medical Management:The management included iron supplementation, dexamethasone, and nebulization with ventolin, as per the consulted teams' recommendations.

Surgical management: Resection and anastomosis surgery

Surgical intervention steps: 1. Under general anesthesia, endotracheal intubation was performed, and the endotracheal tube (ETT) was positioned above the stenotic part of the patient's trachea to ensure airway patency. 2. The patient was then positioned in the left lateral decubitus position for optimal surgical access. 3. A right posterolateral incision was made, the chest was methodically opened in layers, and the right lung was retracted anteriorly to expose the underlying structures. 4. Upon exploration, a dilated supra-carinal tracheal portion and tracheal stenosis approximately 1 cm proximal to the carina were identified. 5. The mediastinal pleura was subsequently opened supero-inferiorly to allow better access to the trachea. 6. The trachea and both main bronchi were secured with nylon tape for control during the procedure. 7. A longitudinal slit incision was then made on the trachea, and the stenotic tracheal portion was resected. 8. Prior to advancing the ETT, an incision was made in the left main bronchus. 9. The ETT was advanced into the left bronchus to maintain ventilation. 10. To facilitate surgical exposure, the right lung was intentionally collapsed. 11. Anastomosis of the trachea was performed using Polydioxanone suture (PDS) 4.0 in an interrupted fashion, both anteriorly and posteriorly. 12. A bubble test was conducted to ensure that there was no air leakage, with negative results confirming the integrity of the anastomosis. 13. A 20 Fr chest tube was then inserted under direct vision to manage any potential pleural effusion or pneumothorax. 14. The chest wall was meticulously closed in layers to ensure the structural integrity of the thoracic cavity. 15. To limit head movement and ensure airway stability postoperatively, PDS 0 was used to secure the patient's chin to the anterior chest wall. The patient was successfully discharged from the ICU on the ninth postoperative day.

Postoperative Course and Management: The patient was admitted to the ICU for meticulous monitoring. To manage increased secretions, the medical team implemented a regimen of regular suction every two hours and chest physiotherapy every three hours. Initially, the patient was successfully extubated on postoperative day 3 but experienced severe respiratory distress, necessitating reintubation the following day. The treatment regimen included antibiotics, corticosteroids, analgesics, and nebulization with 3% hypertonic saline administered every eight hours. Two days after reintubation, the patient was extubated again and continued to receive intermittent continuous positive airway pressure (CPAP), along with ongoing chest physiotherapy, nebulization, corticosteroids, and antibiotics. The chest tube was removed on postoperative day 8, and the patient was subsequently discharged from the ICU on postoperative day 9.

Follow-Up and Further Management: The patient experienced a series of postoperative complications and progressions. On postoperative day 17, she was readmitted to the ICU due to severe respiratory distress, specifically presenting with type 2 respiratory failure accompanied by mixed acidosis. Her condition stabilized, and she was discharged from the ICU on postoperative day 20.

Further evaluations were conducted, and on postoperative day 27, an IV contrast chest CT scan revealed irregular luminal narrowing in the distal trachea measuring 2.4 cm in length and 3.4 mm in width, with the superior caliber extending to 6.3 mm up to the level of the carina. This scan also revealed bilateral left lower and right middle peribronchial opacities, indicative of viral or atypical pneumonia.

Subsequently, the patient was discharged home on postoperative day 32 under the care of a pediatric pulmonologist, who continued to monitor her recovery with further bronchoscope dilation scheduled as part of her ongoing treatment.

This case report describes a rare and complex case of CTS in a 2-year-old female characterized by persistent respiratory distress, stridor and recurrent pneumonia, despite previous interventions for presumed laryngomalacia and asthma. The successful diagnosis and management of this case adds valuable insights to the existing body of literature on isolated CTS, particularly in terms of diagnostic challenges and the efficacy of surgical intervention (30).

CTS is a rare condition with significant clinical variability, making early diagnosis and treatment crucial for preventing severe complications. The estimated incidence of CTS is approximately 1 in 64,500 live births, and it is often associated with other congenital anomalies, predominantly cardiovascular in nature (1,4–6,31–33). This case underscores the diagnostic challenge posed by CTS, especially when it presents with common respiratory symptoms that may be initially attributed to more prevalent pediatric conditions (30,34). The use of an IV contrast chest CT scan played a pivotal role in diagnosing CTS in our patient, aligning with current recommendations that emphasize the importance of detailed imaging in diagnosing and planning treatment for CTS (1,18,29,35).

The literature suggests that the management of CTS requires a tailored, multidisciplinary approach, as demonstrated in our case (36–41). Surgical intervention, specifically resection of the stenotic segment and anastomosis of the short segment, remains the cornerstone of treatment for severe cases of CTS (36,42–46). Our patient's successful outcome, highlighted by surgical correction of tracheal stenosis and comprehensive postoperative care, aligns with findings from previous studies that advocate early surgical intervention for managing CTS to optimize patient prognosis (34,47–49). This finding is consistent with the work of other authors, who emphasized the value of multidisciplinary care and individualized treatment strategies in addressing the complexities of CTS (43,50).

Despite the successful management of this case, it is imperative to acknowledge its limitations. The rarity of CTS and the individualized nature of each patient limit the generalizability of our findings (1,29,51). Moreover, the complexity of CTS management, particularly in patients with concurrent anomalies or those presenting with dysmorphic features, such as our patient, necessitates further research to refine diagnostic and treatment protocols (4,31–33).

Future research should focus on long-term outcomes of surgical interventions for CTS, the development of guidelines for managing complex cases, and the exploration of nonsurgical interventions that could complement the current treatment paradigms (9,28). Additionally, further studies are needed to investigate the genetic underpinnings of CTS, especially in syndromic patients, to enhance our understanding and management of this challenging condition (30,52,53).

In conclusion, this case report highlights the critical role of a meticulous diagnostic process and the need for a multidisciplinary approach for managing CTS. This finding underscores the importance of considering CTS in the differential diagnosis of persistent pediatric respiratory distress and the effectiveness of surgical intervention in improving patient outcomes (29,54). Through this contribution, we aim to enhance awareness and stimulate further research into this rare but impactful condition, with the hope of advancing clinical practice and patient care in pediatric respiratory diseases.

CTS - congenital tracheal stenosis

CT - Computed Tomography

ICU - Intensive Care Unit

ECMO - Extracorporeal membrane oxygenation

MRI - magnetic resonance imaging

MPR - Multiplanar reconstruction

ETT - Endotracheal Tube

SpO2 - Peripheral Oxygen Saturation

PDS - Polydioxanone Suture

CPAP - Continuous Positive Airway Pressure

Ethics Statement and Patient Consent: Ethical approval was waived, and the patient’s parents provided written consent for the publication of this case report.

Conflict of Interest Statement: The authors declare that there are no conflicts of interest regarding the publication of this paper.

Funding Statement: The authors received no specific funding for this work.

Data Availability Statement: The CT scan images utilized in this study are confidential and contain personally identifiable patient information, which is protected under patient privacy regulations. As such, these images are not publicly available. Access to the anonymized data may be granted on a case-by-case basis upon submitting a formal request to the corresponding author. Such requests will be reviewed by the Kenyatta National Hospital's ethics committee to ensure compliance with data protection standards and ethical guidelines. Approval will be contingent upon the requester's adherence to strict confidentiality agreements and the intended use aligning with ethical research practices.

Herrera P, Caldarone C, Forte V, Campisi P, Holtby H, Chait P, et al. The current state of congenital tracheal stenosis. Vol. 23, Pediatric Surgery International. 2007. p. 1033–44.
Hewitt RJ, Butler CR, Maughan EF, Elliott MJ. Congenital tracheobronchial stenosis. Semin Pediatr Surg. 2016 Jun 1;25(3):144–9.
Yokoi A. Congenital tracheal stenosis: what should we look at for successful tracheoplasty? Vol. 7, Translational Pediatrics. AME Publishing Company; 2018. p. 229–32.
Evans JA, Reggin J, Greenberg C. Tracheal agenesis and associated malformations: a comparison with tracheoesophageal fistula and the VACTERL association. Am J Med Genet. 1985;21(1):21–34.
Berdon WE, Baker DH, Wung JT, Chrispin A, Kozlowski K, de Silva M, et al. Complete cartilage-ring tracheal stenosis associated with anomalous left pulmonary artery: the ring-sling complex. Radiology. 1984;152(1):57–64.
Tsuji E, Morita K, Matsuhisa H, Bitoh Y, Hatakeyama T. Incidence of congenital tracheal stenosis in left pulmonary artery sling diagnosed by bronchoscopy. Pediatr Surg Int [Internet]. 2023 Dec 1 [cited 2024 Mar 26];39(1):1–6. Available from: https://link.springer.com/article/10.1007/s00383-023-05527-6
Arunathan R, Ariffin AHZ, Khor KG, Tan SN. Isolated congenital tracheal stenosis: A rare and deadly condition. Pediatr Investig [Internet]. 2019 Sep 1 [cited 2024 Mar 23];3(3):191. Available from:/pmc/articles/PMC7331346/
Benjamin B, Pitkin J, Cohen D. Congenital tracheal stenosis. Ann Otol Rhinol Laryngol [Internet]. 1981 [cited 2024 Mar 25];90(4 Pt 1):364–71. Available from: https://pubmed.ncbi.nlm.nih.gov/7271149/
Antón-Pacheco JL, Cano I, Comas J, Galletti L, Polo L, García A, et al. Management of congenital tracheal stenosis in infancy. European Journal of Cardio-thoracic Surgery. 2006 Jun;29(6):991–6.
Antón-Pacheco JL, Cano I, García A, Martínez A, Cuadros J, Berchi FJ. Patterns of management of congenital tracheal stenosis. J Pediatr Surg. 2003 Oct 1;38(10):1452–8.
Sheikh S, Nagaraj HS, Madden R. Congenital long segment tracheal stenosis presenting with tachypnea. Paediatr Child Health [Internet]. 2000 [cited 2024 Mar 26];5(5):269. Available from:/pmc/articles/PMC2819917/
Schweiger C, Cohen AP, Rutter MJ. Tracheal and bronchial stenoses and other obstructive conditions. J Thorac Dis. 2016;8(11):3369–78.
Extracorporeal Membrane Oxygenation for Perioperative Suppor... : Anesthesia & Analgesia [Internet]. [cited 2024 Mar 23]. Available from: https://journals.lww.com/anesthesia-analgesia/citation/1992/11000/extracorporeal_membrane_oxygenation_for.31.aspx
Walker LK, Wetzel RC, Haller JA. Extracorporeal membrane oxygenation for perioperative support during congenital tracheal stenosis repair. Anesth Analg [Internet]. 1992 [cited 2024 Mar 23];75(5):825–9. Available from: https://pubmed.ncbi.nlm.nih.gov/1416139/
Reis FP Dos, Minamoto H, Bibas BJ, al. et. Treatment of tracheal stenosis with extracorporeal membrane oxygenation support in infants and newborns. Artif Organs. 2021;45:748–53.
Yokoi A. Congenital tracheal stenosis: what should we look at for successful tracheoplasty? Vol. 7, Translational Pediatrics. AME Publishing Company; 2018. p. 229–32.
Elliott M, Roebuck D, Noctor C, McLaren C, Hartley B, Mok Q, et al. The management of congenital tracheal stenosis. Int J Pediatr Otorhinolaryngol. 2003 Dec 1;67(SUPPL. 1):S183–92.
Hernandez RJ, Tucker GF. Congenital tracheal stenosis: Role of CT and high kV films. Pediatr Radiol [Internet]. 1987 Mar [cited 2024 Mar 25];17(3):192–6. Available from: https://link.springer.com/article/10.1007/BF02388158
Speggiorin S, Torre M, Roebuck DJ, Mclaren CA, Elliott MJ. A New Morphologic Classification of Congenital Tracheobronchial Stenosis. ATS. 2012;93:958–61.
Poynot WJ, Gonthier KA, Dunham ME, Crosby TW. Classification of tracheal stenosis in children based on computational aerodynamics. J Biomech. 2020 May 7;104.
Antón-Pacheco JL, Cano I, Comas J, Galletti L, Polo L, García A, et al. Management of congenital tracheal stenosis in infancy. European Journal of Cardio-thoracic Surgery. 2006 Jun;29(6):991–6.
Elliott M, Roebuck D, Noctor C, McLaren C, Hartley B, Mok Q, et al. The management of congenital tracheal stenosis. Int J Pediatr Otorhinolaryngol. 2003;67(SUPPL. 1).
Zibdawi R, El-Andari R, Noga M, Hicks M, Muhieldin M, Phillipos E, et al. Tracheal Reconstruction for Congenital Tracheal Stenosis: A 950-Gram Neonate. Annals of Thoracic Surgery [Internet]. 2022 Oct 1 [cited 2024 Mar 23];114(4):e287–9. Available from: http://www.annalsthoracicsurgery.org/article/S0003497522000431/fulltext
Zhang H, Wang S, Lu Z, Zhu L, Du X, Wang H, et al. Slide tracheoplasty in 81 children: Improved outcomes with modified surgical technique and optimal surgical age. Medicine (United States). 2017 Sep 1;96(38).
Razumovsky AYU, Stepanenko NS, Kulikova N V., Kislenko AA. Thoracoscopic tracheal resection in a child under extracorporeal membrane oxygenation. Pirogov Russian Journal of Surgery. 2024;(1):86–90.
Kocyildirim E, Kanani M, Roebuck D, Wallis C, McLaren C, Noctor C, et al. Long-segment tracheal stenosis: Slide tracheoplasty and a multidisciplinary approach improve outcomes and reduce costs. Journal of Thoracic and Cardiovascular Surgery. 2004;128(6):876–82.
Xu X, Li D, Zhao S, Liu X, Feng Z, Ding H. Treatment of congenital tracheal stenosis by balloon-expandable metallic stents in pediatric intensive care unit. Interact Cardiovasc Thorac Surg. 2012 May;14(5):548–50.
Cheng W, Manson DE, Forte V, Ein SH, MacLusky I, Papsin BC, et al. The role of conservative management in congenital tracheal stenosis: an evidence-based long-term follow-up study. J Pediatr Surg [Internet]. 2006 Jul [cited 2024 Mar 25];41(7):1203–7. Available from: https://pubmed.ncbi.nlm.nih.gov/16818049/
Rimell FL, Stool SE. Diagnosis and Management of Pediatric Tracheal Stenosis. Otolaryngol Clin North Am. 1995 Aug 1;28(4):809–27.
Morais CG, Baptista C, Grilo M, Moreira A, Ribeiro A. Challenging Diagnosis of a Congenital Tracheal Malformation: Considerations From an Intensive Care Perspective. Cureus [Internet]. 2023 Jan 30 [cited 2024 Apr 2];15(1). Available from:/pmc/articles/PMC9978948/
Okamoto T, Nishijima E, Maruo A, Yokoi A, Takamizawa S, Satoh S, et al. Congenital tracheal stenosis: the prognostic significance of associated cardiovascular anomalies and the optimal timing of surgical treatment. J Pediatr Surg [Internet]. 2009 Feb 1 [cited 2024 Mar 26];44(2):325–8. Available from: http://www.jpedsurg.org/article/S0022346808009445/fulltext
Okamoto T, Nishijima E, Maruo A, Yokoi A, Takamizawa S, Satoh S, et al. Congenital tracheal stenosis: the prognostic significance of associated cardiovascular anomalies and the optimal timing of surgical treatment. J Pediatr Surg. 2009 Feb;44(2):325–8.
Sengupta A, Murthy RA. Congenital tracheal stenosis & associated cardiac anomalies: operative management & techniques. J Thorac Dis [Internet]. 2020 Mar 1 [cited 2024 Mar 26];12(3):1184. Available from:/pmc/articles/PMC7139091/
Khan R. The silent storm: a case report of missed antenatal diagnosis leading to fatal respiratory distress and death in a rare case of congenital high airway obstruction syndrome. Int J Contemp Pediatrics [Internet]. 2024 Mar 27;11(4):485–9. Available from: https://www.ijpediatrics.com/index.php/ijcp/article/view/5936
Antón-Pacheco JL, Cano I, Comas J, Galletti L, Polo L, García A, et al. Management of congenital tracheal stenosis in infancy. European Journal of Cardio-thoracic Surgery. 2006 Jun;29(6):991–6.
Kocyildirim E, Kanani M, Roebuck D, Wallis C, McLaren C, Noctor C, et al. Long-segment tracheal stenosis: Slide tracheoplasty and a multidisciplinary approach improve outcomes and reduce costs. Journal of Thoracic and Cardiovascular Surgery. 2004;128(6):876–82.
Doherty C, Neal R, English C, Cooke J, Atkinson D, Bates L, et al. Multidisciplinary guidelines for the management of pediatric tracheostomy emergencies. Anesthesia [Internet]. 2018 Nov 1 [cited 2024 Apr 2];73(11):1400–17. Available from: https://onlinelibrary.wiley.com/doi/full/10.1111/anae.14307
Weitzel NS, Ramakrishnan V, Weyant M, Puskas F. Laryngotracheal stenosis: Clinical manifestations and management. Semin Cardiothorac Vasc Anesth [Internet]. 2011 Dec 22 [cited 2024 Apr 2];15(4):169–75. Available from: https://journals.sagepub.com/doi/10.1177/1089253211427785?icid=int.sj-full-text.similar-articles.9
Chiu P, Kim P. Prognostic factors in the surgical treatment of congenital tracheal stenosis: a multicenter analysis of the literature. J Pediatr Surg. 2006;41:221–5.
Antón-pacheco JL, Comas J V., Luna C, Benavent MI, López M, Ramos V, et al. Treatment strategies in the management of severe complications following slide tracheoplasty in children. European Journal of Cardio-Thoracic Surgery [Internet]. 2014 Aug 1 [cited 2024 Apr 2];46(2):280–5. Available from: https://dx.doi.org/10.1093/ejcts/ezt617
Arcieri L, Giordano R, Murzi B. To do or not to do? The management dilemma of congenital tracheal stenosis in the setting of the ring-sling complex. J Thorac Dis [Internet]. 2017 Dec 1 [cited 2024 Apr 2];9(12):4896–8. Available from: https://jtd.amegroups.org/article/view/17282/html
Rossi ME, Moreddu E, Macé L, Triglia JM, Nicollas R. Surgical management of children presenting with surgical-needed tracheal stenosis. Int J Pediatr Otorhinolaryngol. 2018 May 1;108:219–23.
Ragalie WS, Mitchell ME. Advances in Surgical Treatment of Congenital Airway Disease. Semin Thorac Cardiovasc Surg. 2016 Feb 1;28(1):62–8.
Hewitt RJ, Butler CR, Maughan EF, Elliott MJ. Congenital tracheobronchial stenosis. Semin Pediatr Surg. 2016 Jun 1;25(3):144–9.
Rossi ME, Moreddu E, Macé L, Triglia JM, Nicollas R. Surgical management of children presenting with surgical-needed tracheal stenosis. Int J Pediatr Otorhinolaryngol. 2018 May 1;108:219–23.
Yang JH, Jun TG, Sung K, Jin HC, Young TL, Pyo WP. Repair of Long-segment Congenital Tracheal Stenosis. J Korean Med Sci [Internet]. 2007 Jun 1 [cited 2024 Apr 2];22(3):491–6. Available from: https://doi.org/10.3346/jkms.2007.22.3.491
Backer CL, Holinger LD. A history of pediatric tracheal surgery. World J Pediatr Congenit Heart Surg [Internet]. 2010 Oct [cited 2024 Apr 2];1(3):344–63. Available from: http://www.ncbi.nlm.nih.gov/pubmed/23804890
Jegatheeswaran A, Muthialu N. Commentary: Slide tracheoplasty for congenital tracheal stenosis: Sliding by the missing pieces. Journal of Thoracic and Cardiovascular Surgery [Internet]. 2022 Jun 1 [cited 2024 Apr 2];163(6):2230–1. Available from: http://www.jtcvs.org/article/S0022522321018067/fulltext
Backer C, Mavroudis C, Holinger L. Repair of congenital tracheal stenosis. Semin Thorac Cardiovasc Surg Pediatr Card Surg Annu. 2002;5:173–86.
Antón-Pacheco JL, Cano I, Comas J, Galletti L, Polo L, García A, et al. Management of congenital tracheal stenosis in infancy. European Journal of Cardio-thoracic Surgery. 2006 Jun;29(6):991–6.
Elliott M, Roebuck D, Noctor C, McLaren C, Hartley B, Mok Q, et al. The management of congenital tracheal stenosis. Int J Pediatr Otorhinolaryngol [Internet]. 2003 [cited 2024 Mar 25];67(SUPPL. 1). Available from: https://pubmed.ncbi.nlm.nih.gov/14662192/
Lin LC, Wen WH, Chen PT. Congenital tracheal stenosis in Mowat-Wilson syndrome with nonsense mutation of ZEB2 gene. Pediatr Neonatol [Internet]. 2023 Oct [cited 2024 Apr 2];0(0). Available from: http://www.pediatr-neonatol.com/article/S1875957223001729/fulltext
Saniasiaya J, van der Meer G, Toll EC, McCaffer C, Barber C, Neeff M. Familial congenital laryngotracheal stenosis: A systematic review. Int J Pediatr Otorhinolaryngol. 2024 Feb 1;177:111841.
Ho AS, Koltai PJ. Pediatric Tracheal Stenosis. Otolaryngol Clin North Am. 2008 Oct;41(5):999–1021.

No competing interests reported.

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Isolated Congenital Tracheal Stenosis in a 2-Year-Old Female: A Case Report

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