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Eur J Pediatr Surg 2021; 31(06): 509-517
DOI: 10.1055/s-0040-1719057
Original Article

Surgical Treatment for Asymptomatic Congenital Pulmonary Airway Malformations in Children: Waiting or Not?

Jiahang Zeng
1   Department of Pediatric Surgery, Guangzhou Women and Children's Medical Center, Guangzhou, China
,
Jianhua Liang
1   Department of Pediatric Surgery, Guangzhou Women and Children's Medical Center, Guangzhou, China
,
Le Li
1   Department of Pediatric Surgery, Guangzhou Women and Children's Medical Center, Guangzhou, China
,
Wei Liu
1   Department of Pediatric Surgery, Guangzhou Women and Children's Medical Center, Guangzhou, China
,
Jue Tang
1   Department of Pediatric Surgery, Guangzhou Women and Children's Medical Center, Guangzhou, China
,
Xiaoli Yin
2   College of Life Science, South China Normal University, Guangzhou, China
,
Guocai Yin
3   College of Life Science, Jining Medical University, Jining, Shandong, China
› Author Affiliations Funding This study was funded by Guangzhou Municipal Health and Family Planning Commission (grant number 20191A010022).
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Abstract

Introduction Infection is undoubtedly the most important factor in influencing the timing and surgical strategy of congenital pulmonary airway malformation (CPAM) surgery. However, there have been no studies on the optimal timing of surgery for patients based on the probability of infection. The aim of this study was performed to explore the optimal timing of surgery of CPAM in children from the risk of infection.

Materials and Methods The correlation of age distribution and pulmonary infection of 237 children diagnosed by pathology from January 2012 to January 2020 in Guangzhou Women and Children's Medical Center were analyzed retrospectively. We defined the cases with preoperative computed tomographic findings of infection, pathological findings of large number of neutrophils, tissue cells, and abscess formation as the infection group.

Results The rate of infection in patients less than 2 years old was significantly lower than in patients over 2 years old (11.4% vs. 45.7%, p < 0.001). And the pulmonary lobectomy rate of patients with infection (29.0%) was significantly higher than in noninfected patients (17.2%) and whole patients (36.3%), p = 0.033. Patients with infection lose more blood during surgery (noninfected patients: 81.76 ± 13.14 mL, infected patients: 145.10 ± 25.39 mL, p = 0.027). The univariate analysis revealed that the infection rate of patients over 2 years old was 3.084 times that of patients ≤2 years old (odds ratio [OR]: 3.084, 95% confidence interval [CI]: 1.196–7.954; p = 0.020). The infection rate of CPAM types III and IV patients is lower than types I and II (OR: 0.531, 95% CI: 0.307–0.920; p = 0.024).

Conclusion In consideration of the high risk of infection, lower minimally invasive surgery rate, an increased rate of pulmonary lobectomy, and more blood loss in patients over 2 years old, our study also supports early surgical treatment. Therefore, we suggest that for asymptomatic patients with CPAM I and CPAM II, surgical treatment should be performed when they are less than 2 years old, providing more options for surgical strategies and monitoring of CPAM patients.

Keywords

congenital pulmonary airway malformations - asymptomatic patients - surgical treatment - pulmonary infection

Authors' Contributions

J.Z. and J.L. conceptualized the study and will stand as guarantors. L.L. and W.L. did the literature search. J.Z., J.T., X.Y., and G.Y. collected the data and helped in data analysis. J.Z. drafted the manuscript which underwent a critical appraisal by L.L., W.L., and J.L. All the authors approved the final manuscript and agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.




Publication History

Received: 29 May 2020

Accepted: 21 September 2020

Article published online:
28 February 2021

© 2021. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Bagrodia N, Cassel S, Liao J, Pitcher G, Shilyansky J. Segmental resection for the treatment of congenital pulmonary malformations. J Pediatr Surg 2014; 49 (06) 905-909
    CrossrefPubMedGoogle Scholar
  • 2 Mattioli G, Pio L, Disma NM. et al. Congenital lung malformations: shifting from open to thoracoscopic surgery. Pediatr Neonatol 2016; 57 (06) 463-466
    CrossrefPubMedGoogle Scholar
  • 3 Berrocal T, Madrid C, Novo S, Gutiérrez J, Arjonilla A, Gómez-León N. Congenital anomalies of the tracheobronchial tree, lung, and mediastinum: embryology, radiology, and pathology. Radiographics 2004; 24 (01) e17
    CrossrefPubMedGoogle Scholar
  • 4 Mehta RP, Faquin WC, Cunningham MJ. Cervical bronchogenic cysts: a consideration in the differential diagnosis of pediatric cervical cystic masses. Int J Pediatr Otorhinolaryngol 2004; 68 (05) 563-568
    CrossrefPubMedGoogle Scholar
  • 5 Kotecha S, Barbato A, Bush A. et al. Antenatal and postnatal management of congenital cystic adenomatoid malformation. Paediatr Respir Rev 2012; 13 (03) 162-170
    CrossrefPubMedGoogle Scholar
  • 6 Stocker LJ, Wellesley DG, Stanton MP, Parasuraman R, Howe DT. The increasing incidence of foetal echogenic congenital lung malformations: an observational study. Prenat Diagn 2015; 35 (02) 148-153
    CrossrefPubMedGoogle Scholar
  • 7 Tocchioni F, Lombardi E, Ghionzoli M, Ciardini E, Noccioli B, Messineo A. Long-term lung function in children following lobectomy for congenital lung malformation. J Pediatr Surg 2017; 52 (12) 1891-1897
    CrossrefPubMedGoogle Scholar
  • 8 Durell J, Thakkar H, Gould S, Fowler D, Lakhoo K. Pathology of asymptomatic, prenatally diagnosed cystic lung malformations. J Pediatr Surg 2016; 51 (02) 231-235
    CrossrefPubMedGoogle Scholar
  • 9 Lakhoo K. Management of congenital cystic adenomatous malformations of the lung. Arch Dis Child Fetal Neonatal Ed 2009; 94 (01) F73-F76
    CrossrefPubMedGoogle Scholar
  • 10 Lau CT, Kan A, Shek N, Tam P, Wong KK. Is congenital pulmonary airway malformation really a rare disease? Result of a prospective registry with universal antenatal screening program. Pediatr Surg Int 2017; 33 (01) 105-108
    CrossrefPubMedGoogle Scholar
  • 11 Parikh DH, Rasiah SV. Congenital lung lesions: postnatal management and outcome. Semin Pediatr Surg 2015; 24 (04) 160-167
    CrossrefPubMedGoogle Scholar
  • 12 Ruchonnet-Metrailler I, Leroy-Terquem E, Stirnemann J. et al. Neonatal outcomes of prenatally diagnosed congenital pulmonary malformations. Pediatrics 2014; 133 (05) e1285-e1291
    CrossrefPubMedGoogle Scholar
  • 13 Stanton M. The argument for a non-operative approach to asymptomatic lung lesions. Semin Pediatr Surg 2015; 24 (04) 183-186
    CrossrefPubMedGoogle Scholar
  • 14 Singh R, Davenport M. The argument for operative approach to asymptomatic lung lesions. Semin Pediatr Surg 2015; 24 (04) 187-195
    CrossrefPubMedGoogle Scholar
  • 15 Papagiannopoulos K, Hughes S, Nicholson AG, Goldstraw P. Cystic lung lesions in the pediatric and adult population: surgical experience at the Brompton Hospital. Ann Thorac Surg 2002; 73 (05) 1594-1598
    CrossrefPubMedGoogle Scholar
  • 16 McBride JT, Wohl ME, Strieder DJ. et al. Lung growth and airway function after lobectomy in infancy for congenital lobar emphysema. J Clin Invest 1980; 66 (05) 962-970
    CrossrefPubMedGoogle Scholar
  • 17 Nakajima C, Kijimoto C, Yokoyama Y. et al. Longitudinal follow-up of pulmonary function after lobectomy in childhood - factors affecting lung growth. Pediatr Surg Int 1998; 13 (5-6): 341-345
    CrossrefPubMedGoogle Scholar
  • 18 Calvert JK, Lakhoo K. Antenatally suspected congenital cystic adenomatoid malformation of the lung: postnatal investigation and timing of surgery. J Pediatr Surg 2007; 42 (02) 411-414
    CrossrefPubMedGoogle Scholar
  • 19 Criss CN, Musili N, Matusko N, Baker S, Geiger JD, Kunisaki SM. Asymptomatic congenital lung malformations: is nonoperative management a viable alternative?. J Pediatr Surg 2018; 53 (06) 1092-1097
    CrossrefPubMedGoogle Scholar
  • 20 Nasr A, Bass J. Thoracoscopic vs open resection of congenital lung lesions: a meta-analysis. J Pediatr Surg 2012; 47 (05) 857-861
    CrossrefPubMedGoogle Scholar
  • 21 Kunisaki SM, Powelson IA, Haydar B. et al. Thoracoscopic vs open lobectomy in infants and young children with congenital lung malformations. J Am Coll Surg 2014; 218 (02) 261-270
    CrossrefPubMedGoogle Scholar
  • 22 Kim HK, Choi YS, Kim K. et al. Treatment of congenital cystic adenomatoid malformation: should lobectomy always be performed?. Ann Thorac Surg 2008; 86 (01) 249-253
    CrossrefPubMedGoogle Scholar
  • 23 Fascetti-Leon F, Gobbi D, Pavia SV. et al. Sparing-lung surgery for the treatment of congenital lung malformations. J Pediatr Surg 2013; 48 (07) 1476-1480
    CrossrefPubMedGoogle Scholar
  • 24 Johnson SM, Grace N, Edwards MJ, Woo R, Puapong D. Thoracoscopic segmentectomy for treatment of congenital lung malformations. J Pediatr Surg 2011; 46 (12) 2265-2269
    CrossrefPubMedGoogle Scholar
  • 25 Jelin EB, O'Hare EM, Jancelewicz T, Nasr I, Boss E, Rhee DS. Optimal timing for elective resection of asymptomatic congenital pulmonary airway malformations. J Pediatr Surg 2018; 53 (05) 1001-1005
    CrossrefPubMedGoogle Scholar
  • 26 Gulack BC, Leraas HJ, Ezekian B. et al. Outcomes following elective resection of congenital pulmonary airway malformations are equivalent after 3 months of age and a weight of 5 kg. J Pediatr Surg 2017; 17: 30639-30645
    PubMedGoogle Scholar
  • 27 Makhijani AV, Wong FY. Conservative post-natal management of antenatally diagnosed congenital pulmonary airway malformations. J Paediatr Child Health 2018; 54 (03) 267-271
    CrossrefPubMedGoogle Scholar
  • 28 Style CC, Cass DL, Verla MA. et al. Early vs late resection of asymptomatic congenital lung malformations. J Pediatr Surg 2019; 54 (01) 70-74
    CrossrefPubMedGoogle Scholar
  • 29 Bush A. Prenatal presentation and postnatal management of congenital thoracic malformations. Early Hum Dev 2009; 85 (11) 679-684
    CrossrefPubMedGoogle Scholar
  • 30 Ueda K, Gruppo R, Unger F, Martin L, Bove K. Rhabdomyosarcoma of lung arising in congenital cystic adenomatoid malformation. Cancer 1977; 40 (01) 383-388
    CrossrefPubMedGoogle Scholar
  • 31 Benjamin DR, Cahill JL. Bronchioloalveolar carcinoma of the lung and congenital cystic adenomatoid malformation. Am J Clin Pathol 1991; 95 (06) 889-892
    CrossrefPubMedGoogle Scholar
  • 32 Miller JA, Corteville JE, Langer JC. Congenital cystic adenomatoid malformation in the fetus: natural history and predictors of outcome. J Pediatr Surg 1996; 31 (06) 805-808
    CrossrefPubMedGoogle Scholar
  • 33 Puligandla PS, Laberge JM. Congenital lung lesions. Clin Perinatol 2012; 39 (02) 331-347
    CrossrefPubMedGoogle Scholar