Evaluation of the Respiratory Impact After Conventional or Minimally Invasive Esophageal Atresia Surgery (RestriMIS)

Evaluation of the Respiratory Impact of Post-operative Chest Wall Anomalies After Conventional or Minimally Invasive Esophageal Atresia Surgery

Right thoracotomy, conventional approach to esophageal atresia repair, leads to up to 60% radiological chest wall sequelae anomalies. The impact of these anomalies on the patient's respiratory function remains unknown. Minimally invasive thoracic surgery considerably reduces this rate.

The primary objective of this study is to assess the occurrence of restrictive lung disease in patients with type III esophageal atresia depending on the type of surgical approach (Conventional or minimally invasive).

The primary endpoint will be he occurrence of restrictive lung disease , objectified by pulmonary function tests (PFTs), carried out according to the current national guidelines (PNDS = protocole national de diagnostic et de soins).

Study Overview

• Status: Unknown
• Conditions: Esophageal Atresia; Restrictive Lung Disease; Oesophageal Atresia; Chest Wall Anomaly

Detailed Description

Right thoracotomy, conventional approach to esophageal atresia repair, leads to up to 60% radiological chest wall sequelae anomalies. The impact of these anomalies on the patient's respiratory function remains unknown. Minimally invasive thoracic surgery considerably reduces this rate.

The primary objective of this study is to assess the occurrence of restrictive lung disease in patients with type III esophageal atresia depending on the type of surgical approach (Conventional or minimally invasive).

The primary endpoint will be the occurrence of restrictive lung disease, as assessed by pulmonary function tests (PFTs), carried out according to the current national guidelines (PNDS = protocole national de diagnostic et de soins).

The secondary endpoints will be to measure the severity of the restrictive disease, to look for other respiratory alterations, to correlate radiological chest wall sequelae anomalies with the impact on respiratory function and to look for a causal relationship between the surgical technique used and the respiratory impact.

The methodology used will be a retrospective non interventional study on the cohort of patients included in the national esophageal atresia registry (CRACMO, Lille University Hospital) between the 1st of january 2008 and the 31st of December 2013.

All the patients included in the national esophageal atresia registry (CRACMO) having had an operation for type III esophageal atresia (long gap esophageal atresia excluded), as defined by the Ladd Classification, will be included in this study.

The exclusion criterion will be patients lost to follow up or deceased, patients having had no pulmonary function tests (PFTs) or no thoracic X-Ray during the first 6 to 9 years of follow up and patients having had thoracic surgery before the esophageal atresia repair.

The number of patients expected in the national esophageal atresia registry over the 6 years excedes 500. The number of thoracoscopy repairs should be about 50.

This study should allow us to determine if minimally invasive surgery is beneficial on mid-term respiratory function in children, related to possible post-operative chest wall sequelae.

The results obtained from this study should lead to recommendations concerning the surgical approach to esophageal atresia repair to improve the prognosis of chest wall anomalies and respiratory function in these patients. It should also help to identify patient subgroups which would benefit from a reinforced respiratory follow up. This could then lead to a hospital clinical research program (PHRC)

• Study Type: Observational
• Enrollment (Anticipated): 500

Contacts and Locations

Study Locations

• France
  • Lille, France, 59000
    • CRACMO - centre de référence des atrésies de l'oesophage

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: ADULT; OLDER_ADULT; CHILD
• Accepts Healthy Volunteers: No
• Genders Eligible for Study: All

• Sampling Method: Non-Probability Sample

Study Population

All the patients included in the national esophageal atresia registry (CRACMO) having had an operation for type III esophageal atresia, as defined by the Ladd classification between 01/01/2008 and 31/12/2013 and being followed up with thoracic X-rays and PFTs.

Description

Inclusion Criteria:

• Patients included in the national esophageal atresia registry (CRACMO)
• Operation for type III esophageal atresia (Ladd classification)
• Between 01/01/2008 and 31/12/2013.

Exclusion Criteria:

• Long gap esophageal atresia
• Patients lost to follow up
• Deceased
• No PFTs or X-rays between 6 and 9 years of follow up
• Patients having had thoracic surgery before the esophageal atresia repair

Study Plan

How is the study designed?

Number of groups / cohorts

2

Cohorts and Interventions

Group / Cohort
Conventional surgery; Patients having had esophageal atresia (type III, long gap excluded) repair by conventional surgery (right thoracotomy) or patients having had minimally invasive surgery converted to thoracotomy between the 1st of january 2008 and the 31st of December 2013 and registered on the national esophageal atresia registry (CRACMO, Lille university hospital)
Minimally invasive surgery; Patients having had esophageal atresia (type III, long gap excluded) repair through minimally invasive surgery between the 1st of january 2008 and the 31st of December 2013 and registered on the national esophageal atresia registry (CRACMO, Lille university hospital)

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
To assess the occurrence of restrictive lung disease in patients with type III esophageal atresia depending on the type of surgical approach (Conventional or minimally invasive).	Objectified by pulmonary function tests (PFTs), carried out according to the current national guidelines. Restrictive lung disease defined by: FEV1/FVC ratio > -1.64 Z-score and CVF < -1.64 Z-score according to ATS/ERS-GLI (American Thoracic Society & European Respiratory Society - Global Lungs Initiative) recommendations.	6 to 9 years of age.

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Severity of restrictive lung disease	Depending on Z-score value	6 to 9 years of age
Assesse the occurrence of obstructive or mixed lung disease	Objectified by pulmonary function tests (PFTs), carried out according to the current national guidelines. Mixed lung disease defined by: FEV1/FVC ratio < -1.64 Z-score and CVF < -1.64 Z-score and obstructive lung disease	6 to 9 years
Mortality rate	Percentage of mortality in each group, cause of death linked directly to surgery or not	Time of surgery to 6 to 9 years consultation
Percentage of post-operative complications depending on the type of surgery	Bleeding, infection, anastomotic stenosis, anastomotic leak	Time of surgery to 6 to 9 years consultation
Chest wall anomalies detected on thoracic X-rays	Hemivertebra, rib fusion, intercostal abnormalities, scoliosis	6 to 9 years of age
Correlation between post-operative chest wall anomalies and restrictive lung disease	In each group, comparison of the percentage of chest wall anomalies detected on the X-rays and the percentage of restrictive lung disease cases	6 to 9 years of age

Collaborators and Investigators

• Sponsor: University Hospital, Angers
• Collaborators: Filière des Maladies Rares Abdomino-THOraciques : FIMATHO; Institut de Recherche en Santé, Environnement et le Travail, France

Publications and helpful links

General Publications

• Bastard F, Bonnard A, Rousseau V, Gelas T, Michaud L, Irtan S, Piolat C, Ranke-Chretien A, Becmeur F, Dariel A, Lamireau T, Petit T, Fouquet V, Le Mandat A, Lefebvre F, Allal H, Borgnon J, Boubnova J, Habonimana E, Panait N, Buisson P, Margaryan M, Michel JL, Gaudin J, Lardy H, Auber F, Borderon C, De Vries P, Jaby O, Fourcade L, Lecompte JF, Tolg C, Delorme B, Schmitt F, Podevin G. Thoracic skeletal anomalies following surgical treatment of esophageal atresia. Lessons from a national cohort. J Pediatr Surg. 2018 Apr;53(4):605-609. doi: 10.1016/j.jpedsurg.2017.07.013. Epub 2017 Jul 21.

Study record dates

Study Major Dates

• Study Start (ANTICIPATED): January 1, 2020
• Primary Completion (ANTICIPATED): August 1, 2020
• Study Completion (ANTICIPATED): December 1, 2020

Study Registration Dates

• First Submitted: October 21, 2019
• First Submitted That Met QC Criteria: October 22, 2019
• First Posted (ACTUAL): October 23, 2019

Study Record Updates

• Last Update Posted (ACTUAL): December 20, 2019
• Last Update Submitted That Met QC Criteria: December 19, 2019
• Last Verified: December 1, 2019

More Information

Terms related to this study

• Keywords: scoliosis; thoracoscopy; Respiratory function; esophageal atresia; rib fusion
• Additional Relevant MeSH Terms: Digestive System Diseases; Respiratory Tract Diseases; Gastrointestinal Diseases; Esophageal Diseases; Digestive System Abnormalities; Lung Diseases; Congenital Abnormalities; Esophageal Atresia
• Other Study ID Numbers: 49RC19_0185

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No