Celine Delestrain1, Naziha Khen-Dunlop2,3, Alice Hadchouel1,3, Pierrick Cros4, Héloïse Ducoin5, Michael Fayon6, Isabelle Gibertini7, André Labbé8, Géraldine Labouret9, Marie-Noëlle Lebras10, Guillaume Lezmi1,3,11, Fouad Madhi11,12, Guillaume Thouvenin11,13, Caroline Thumerelle14, Christophe Delacourt15,3,11. 1. Pneumologie Pédiatrique. 2. Chirurgie Pédiatrique, Hôpital Necker-Enfants Malades, Assistance Publique-Hospitaux de Paris, Paris, France. 3. Université Paris-Descartes, Paris, France. 4. Pneumologie Pédiatrique, Centre Hospitalier Régional et Universitaire de Brest, Brest, France. 5. Pneumologie Pédiatrique, Centre Hospitalier de Lens, Lens, France. 6. Pneumologie Pédiatrique, Centre Hospitalier Universitaire de Bordeaux, Bordeaux, France. 7. Pneumologie Pédiatrique, Centre Hospitalier Universitaire de Tours, Tours, France. 8. Pneumologie Pédiatrique, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France. 9. Pneumologie Pédiatrique, Centre Hospitalier Universitaire de Toulouse, Toulouse, France. 10. Pneumologie Pédiatrique, Hôpital Universitaire Robert Debré, Assistance Publique-Hospitaux de Paris, Paris, France. 11. Centre de Référence des Maladies Respiratoires Rares, Paris, France. 12. Pneumologie Pédiatrique, Centre Hospitalier Intercommunal Créteil,Créteil, France. 13. Pneumologie Pédiatrique, Hôpital Armand Trousseau, Assistance Publique Hospitaux de Paris, Paris, France; and. 14. Pneumologie Pédiatrique, Centre Hospitalier Régional Universitaire de Lille, Lille, France. 15. Pneumologie Pédiatrique, christophe.delacourt@nck.aphp.fr.
Abstract
BACKGROUND AND OBJECTIVES: The actual frequency of respiratory symptoms related to congenital pulmonary malformations (CPMs) remains undetermined. The goal of this study was to prospectively evaluate the respiratory symptoms occurring in infants with prenatally diagnosed CPMs, identify factors associated with the occurrence of these symptoms, and evaluate their resolution after surgery. METHODS: Infectious and noninfectious respiratory symptoms were prospectively collected in a French multicenter cohort of children with CPMs. RESULTS: Eighty-five children were followed up to the mean age of 2.1 ± 0.4 years. Six children (7%) underwent surgery during the first 28 days of life. Of the 79 remaining children, 33 (42%) had respiratory symptoms during infancy before any surgery. Wheezing was the dominant symptom (24 of 79 [30%]), and only 1 infant had documented infection of the cystic lobe. Symptoms were more frequent in children with noncystic CPMs, prenatally (P = .01) or postnatally (P < .03), and with postnatally hyperlucent CPMs (P < .01). Sixty-six children underwent surgery during the follow-up period, and 40% of them displayed symptoms after the intervention. Six children had documented pneumonia during the postoperative period. At the end of the follow-up, pectus excavatum was observed in 10 children, significantly associated with thoracotomy (P < .02) or with surgery before the age of 6 months (P < .002). CONCLUSIONS: CPMs are frequently associated with wheezing episodes. Surgery had no significant impact on these symptoms but was associated with a paradoxical increase in pulmonary infections, as well as an increased risk of pectus excavatum after thoracotomy.
BACKGROUND AND OBJECTIVES: The actual frequency of respiratory symptoms related to congenital pulmonary malformations (CPMs) remains undetermined. The goal of this study was to prospectively evaluate the respiratory symptoms occurring in infants with prenatally diagnosed CPMs, identify factors associated with the occurrence of these symptoms, and evaluate their resolution after surgery. METHODS: Infectious and noninfectious respiratory symptoms were prospectively collected in a French multicenter cohort of children with CPMs. RESULTS: Eighty-five children were followed up to the mean age of 2.1 ± 0.4 years. Six children (7%) underwent surgery during the first 28 days of life. Of the 79 remaining children, 33 (42%) had respiratory symptoms during infancy before any surgery. Wheezing was the dominant symptom (24 of 79 [30%]), and only 1 infant had documented infection of the cystic lobe. Symptoms were more frequent in children with noncystic CPMs, prenatally (P = .01) or postnatally (P < .03), and with postnatally hyperlucent CPMs (P < .01). Sixty-six children underwent surgery during the follow-up period, and 40% of them displayed symptoms after the intervention. Six children had documented pneumonia during the postoperative period. At the end of the follow-up, pectus excavatum was observed in 10 children, significantly associated with thoracotomy (P < .02) or with surgery before the age of 6 months (P < .002). CONCLUSIONS: CPMs are frequently associated with wheezing episodes. Surgery had no significant impact on these symptoms but was associated with a paradoxical increase in pulmonary infections, as well as an increased risk of pectus excavatum after thoracotomy.
Authors: Annelieke Hijkoop; Marloes M van Schoonhoven; Joost van Rosmalen; Dick Tibboel; Monique H M van der Cammen-van Zijp; Mariëlle W Pijnenburg; Titia E Cohen-Overbeek; Johannes M Schnater; Hanneke IJsselstijn Journal: Pediatr Pulmonol Date: 2019-04-22