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Oral Presentation - 31
Key factors linked to the development of restrictive lung syndrome in children following surgery for type III esophageal atresia.
Jeanne Goulin 1, Thomas Brigly 2, François Bastard 1, Arnaud Bonnard 3, Véronique Rousseau 4, Thomas Gelas 5, Audrey Guinot 6, Edouard Habonimana 7, Anne Breton 8, Aline Ranke 9, Isabelle Talon 10, Sabine Irtan 11, Thierry Lamireau 12, Catherine Jacquier 13, Frédéric Elbaz 14, Nicolas Kalfa 15, Nicoleta Panait 16, Virginie Fouquet 17, Hubert Lardy 18, Aurélien Scalabre 19, Philippe Buisson 20, Marc Margaryan 21, Frédéric Auber 22, Céline Grosos 23, Corinne Borderon 24, Cécilia Tölg 25, Guillaume Podevin 1, Rony Sfeir 26, Frédéric Gottrand 26, Françoise Schmitt 1
1 Angers university Hospital Centre
2 Laval Regional Hospital Centre
3 AP-HP Robert Debré Hospital
4 AP-HP Necker Sick Children Hospital
5 Lyon University Hospital Centre
6 Nantes University Hospital Centre
7 Rennes University Hospital Centre
8 Toulouse University Hospital Centre
9 Nancy University Hospital Centre
10 Hautepierre University Hospital of Strasbourg
11 AP-HP Armand Trousseau Hospital
12 Bordeaux University Hospital Centre
13 Grenoble University Hospital Centre
14 Rouen University Hospital Centre
15 Montpellier University Hospital Centre
16 AP-HM University Hospital Centre Marseille
17 AP-HP Bicêtre Hospital
18 Tours University Hospital Centre
19 Saint Etienne University Hospital Centre
20 Amiens University Hospital Centre
21 Le Mans Regional Hospital
22 Besançon University Hospital Centre
23 Limoges University Hospital Centre
24 Clermont-Ferrant University Hospital Centre
25 Martinique University Hospital Centre
26 Lille University Hospital
Purpose: To evaluate the incidence of restrictive lung syndrome in children after type III esophageal atresia (EA) surgery and to identify potential associated factors.
Method: After local institution review board approval, a multicenter retrospective study was conducted using data from 216 patients with type III EA included in a population-based registry between 2008 and 2013. Restrictive lung syndrome was defined based on the Z-score of forced vital capacity measured during pulmonary function tests conducted between 6 and 9 years of age. (ClinicalTrial NCT04136795)
Results: At follow-up (7.5 ± 1.5 years), 58 (27%) had pure restrictive syndrome and 137 (63%) had normal respiratory function. Patients with restrictive syndrome were more often premature (52% vs. 30%, p = 0.0005) and had more associated cardiac malformations (36% vs. 12%, p = 0.0003). Surgically, these patients more frequently required delayed anastomosis (8.6% vs. 1.5%, p = 0.03), gastrostomy (17% vs. 1.5%, p = 0.0001), or other thoracic surgeries (17% vs. 5%, p = 0.01). At age 6, they had a lower body mass index and more respiratory history (85% vs. 63%, p = 0.002) but no significant increase in thoracic wall sequelae (73% vs. 69%, p = 0.84). Multivariate analysis showed that restrictive syndrome was associated with Caucasian origins (Odds ratio OR = 4.3 [1.2 – 15.4]), a history of tracheomalacia (OR 4.06 [1.62-10.20]), cardiac anomalies (OR 5.75 [1.95-16.95]), and gastroesophageal reflux (OR 3.06 [1.26-7.41]). On the contrary, neither surgical factors nor chest-wall sequelae were associated with a restrictive pattern.
Conclusion: These findings suggest that restrictive lung syndrome affects more than a quater of children with type III EA and is linked to patient-associated anomalies rather than surgical or health care-associated factors.