Congenital Lung Malformations: Outcomes after Minimally Invasive and Open Surgery in Infancy

Abstract

Background

The management of congenital lung malformations (CLM) remains controversial. The aim of this study was to analyze the timing, approach, and outcomes of surgical treatment in children with CLM.

Patients and Methods

A cohort study was conducted comparing children with CLM who underwent thoracoscopic resection with open resection via thoracotomy. All children were treated according to an institutionalized multidisciplinary coordinated treatment algorithm.

Results

Between 2002 and 2019, 68 children with CLM were treated. Thoracoscopic resection of CLM (n=44) resulted in a statistically significant longer operative time (mean 169 vs. 97 minutes, p=0.04) but shorter hospital stay (mean 7.2 vs. 16.7/12 days, p=0.01) compared to thoracotomy (n=15) or after conversion to thoracotomy (n=9). There were no major complications in either group. At a mean follow-up of 32.2 months (range 0.5–163), patients after thoracoscopic resection had statistically significantly fewer rip fusions than patients after open resection (n=0 vs. 2/2, p≤0.004) and less chest wall asymmetry than after conversion (n=0 vs. 2, p=0.004).

Discussion

Thoracotomy proved to be a fast and safe surgical approach in respiratory unstable CLM patients. In respiratory stable CLM patients, thoracoscopic resection was feasible in early infancy with good surgical and musculoskeletal outcomes.

Conclusion

Thoracoscopic resection of CLM offers advantages over resection by thoracotomy in terms of surgical recovery and musculoskeletal function. Therefore, it should be offered to respiratory-stable patients in specialized centers.

Zusammenfassung

Hintergrund

Die Behandlung von kongenitalen Lungenfehlbildungen (CLM) ist nach wie vor umstritten. Ziel dieser Studie war es, den Zeitpunkt, das Operationsverfahren und die Ergebnisse bei Kindern mit CLM zu analysieren.

Patienten und Methoden

In einer Kohortenstudie wurden Kinder mit CLM nach thorakoskopischer Resektion mit Kindern nach offener Resektion mittels Thorakotomie verglichen. Alle Kinder wurden nach einem interdisziplinär abgestimmten Therapieplan behandelt.

Ergebnisse

Zwischen 2002 und 2019 wurden 68 Kinder mit CLM operiert. Die thorakoskopische Resektion der CLM (n=44) führte zu einer statistisch signifikant längeren Operationszeit (Mittelwert 169 vs. 97 Minuten, p=0,04), aber zu einem kürzeren Krankenhausaufenthalt (Mittelwert 7,2 vs. 16,7/12 Tage, p=0,01) im Vergleich zur Thorakotomie (n=15) bzw. nach Konversion zur Thorakotomie (n=9). Schwerwiegende Komplikationen traten in keiner Gruppe auf. Bei einer mittleren Nachbeobachtungszeit von 32,2 Monaten (0,5–163) hatten Patienten nach thorakoskopischer Resektion statistisch signifikant weniger Rippenfusionen als Patienten nach offener Resektion (n=0 vs. 2/2, p≤0,004) und weniger Brustwandasymmetrien als Patienten nach Konversion (n=0 vs. 2, p=0,004).

Diskussion

Die Thorakotomie erwies sich als schnelles und sicheres Operationsverfahren bei respiratorisch instabilen CLM-Patienten. Bei respiratorisch stabilen CLM-Patienten war die thorakoskopische Resektion bereits im frühen Säuglingsalter mit guten chirurgischen und muskuloskelettalen Ergebnissen durchführbar.

Schlussfolgerung

Die thorakoskopische Resektion der CLM bietet im Vergleich zur Resektion mittels Thorakotomie Vorteile hinsichtlich der Rekonvaleszenz der muskuloskelettalen Funktion. Sie sollte daher respiratorisch stabilen Patienten in spezialisierten Zentren angeboten werden.

Keywords

congenital lung malformations - infancy - asymptomatic - symptomatic - thoracoscopy - thoracotomy

Schlüsselwörter

Angeborene Lungenfehlbildungen - Säuglingsalter - Asymptomatisch - Symptomatisch - Thorakoskopie - Thorakotomie

Publication History

Article published online:
07 February 2025

• References

• 1 Kirschner H-J, Fuchs J. Angeborene pulmonale Malformationen. Zentralbl Chir 2017; 142: 127-139
• 2 Stocker LJ, Wellesley DG, Stanton MP. et al. The increasing incidence of foetal echogenic congenital lung malformations: an observational study. Prenatal Diagnosis 2015; 35: 148-153
• 3 Lau CT, Kan A, Shek N. et al. Is congenital pulmonary airway malformation really a rare disease? Result of a prospective registry with universal antenatal screening program. Pediatr Surg Int 2017; 33: 105-108
• 4 Kunisaki SM, Saito JM, Fallat ME. et al. Fetal Risk Stratification and Outcomes in Children with Prenatally Diagnosed Lung Malformations : Results from a Multi-Institutional Research Collaborative. Annals of Surgery 2022; 276: e622-e630
• 5 Stanton M. The argument for a non-operative approach to asymptomatic lung lesions. Seminars in Pediatric Surgery 2015; 24: 183-186
• 6 Singh R, Davenport M. The argument for operative approach to asymptomatic lung lesions. Seminars in Pediatric Surgery 2015; 24: 187-195
• 7 Morini F, Zani A, Conforti A. et al. Current Management of Congenital Pulmonary Airway Malformations: A “European Pediatric Surgeons’ Association” Survey. Eur J Pediatr Surg 2018; 28: 1-5
• 8 Lieber J, Urla CI, Baden W. et al. Experiences and challenges of thorcoscopic lung surgery in the pediatric age group. International Journal of Surgery 2015; 23: 169-175
• 9 Kirschner H-J, Fuchs J. Erfahrungen mit der minimalinvasiven anatomischen Lungenresektion im Kindesalter. In: pädiatrische praxis. Mediengruppe Oberfranken – Fachverlage GmbH & Co. KG; 2017: 281-290
• 10 Fuchs J. Congenital Lung Malformations. In: Parikh D, Rajesh PB, Hrsg. Tips and Tricks in Thoracic Surgery. London: Springer; 2018: 3-16
• 11 Neunhoeffer F, Warmann SW, Hofbeck M. et al. Elevated intrathoracic CO2 pressure during thoracoscopic surgery decreases regional cerebral oxygen saturation in neonates and infants – A pilot study. Pediatric Anesthesia 2017; 27: 752-759
• 12 Dindo D, Demartines N, Clavien P-A. Classification of Surgical Complications: A New Proposal With Evaluation in a Cohort of 6336 Patients and Results of a Survey. Annals of Surgery 2004; 240: 205
• 13 Rizzo F, Torre M, Guerriero V. et al. Congenital Lung Malformations. In: Mattioli G, Petralia P, Torre M, Hrsg. Pediatric Thoracic Surgery. Cham: Springer International Publishing; 2021: 149-159
• 14 Polites SF, Habermann EB, Zarroug AE. et al. Thoracoscopic Vs open resection of congenital cystic lung disease- utilization and outcomes in 1120 children in the United States. Journal of Pediatric Surgery 2016; 51: 1101-1105
• 15 Weller JH, Peter SDST, Fallat ME. et al. Thoracoscopic versus open lobectomy in infants with congenital lung malformations: A multi-institutional propensity score analysis. Journal of Pediatric Surgery 2021; 56: 2148-2156
• 16 Laje P, Pearson EG, Simpao AF. et al. The first 100 infant thoracoscopic lobectomies: Observations through the learning curve and comparison to open lobectomy. Journal of Pediatric Surgery 2015; 50: 1811-1816
• 17 Mon RA, Johnson KN, Ladino-Torres M. et al. Diagnostic accuracy of imaging studies in congenital lung malformations. Archives of Disease in Childhood – Fetal and Neonatal Edition 2019; 104: F372-F377
• 18 Kellenberger CJ, Amaxopoulou C, Moehrlen U. et al. Structural and perfusion magnetic resonance imaging of congenital lung malformations. Pediatr Radiol 2020; 50: 1083-1094
• 19 Lacher M, Kuebler JF, Dingemann J. et al. Minimal invasive surgery in the newborn: Current status and evidence. Seminars in Pediatric Surgery 2014; 23: 249-256
• 20 Kulaylat AN, Engbrecht BW, Hollenbeak CS. et al. Comparing 30-day outcomes between thoracoscopic and open approaches for resection of pediatric congenital lung malformations: Evidence from NSQIP. Journal of Pediatric Surgery 2015; 50: 1716-1721
• 21 Kunisaki SM, Saito JM, Fallat ME. et al. Development of a multi-institutional registry for children with operative congenital lung malformations. Journal of Pediatric Surgery 2020; 55: 1313-1318
• 22 Engall N, Sethuraman C, Wilkinson DJ. et al. Does Timing of Resection Influence the Presence of Inflammation within Congenital Lung Malformations?. Eur J Pediatr Surg 2023; 33: 81-84
• 23 Lau C-T, Wong KKY. Long-term pulmonary function after lobectomy for congenital pulmonary airway malformation: is thoracoscopic approach really better than open?. Journal of Pediatric Surgery 2018; 53: 2383-2385
• 24 Jelin EB, O’Hare EM, Jancelewicz T. et al. Optimal timing for elective resection of asymptomatic congenital pulmonary airway malformations. Journal of Pediatric Surgery 2018; 53: 1001-1005
• 25 Feinberg A, Hall NJ, Williams GM. et al. Can congenital pulmonary airway malformation be distinguished from Type I pleuropulmonary blastoma based on clinical and radiological features?. Journal of Pediatric Surgery 2016; 51: 33-37