PDF herunterladen
Eur J Pediatr Surg 2024; 34(01): 063-068
DOI: 10.1055/a-2130-2564
Original Article

Respiratory and Musculoskeletal Long-Term Outcomes after Surgical Resection of Congenital Cystic Adenomatoid Malformation of the Lung in Newborns, Infants, and Toddlers

Matteo Busti
1   Newborn Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
2   Department of Systems Medicine, University of “Tor Vergata,” Rome, Italy
,
Angelo Zarfati 
1   Newborn Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
2   Department of Systems Medicine, University of “Tor Vergata,” Rome, Italy
,
Laura Valfre
1   Newborn Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
,
Andrea Conforti
1   Newborn Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
,
Pietro Bagolan
1   Newborn Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
2   Department of Systems Medicine, University of “Tor Vergata,” Rome, Italy
› Institutsangaben
› Weitere Informationen
Auch verfügbar auf
    Lizenzen und Reprints

Abstract

Introduction The long-term outcomes of children who underwent surgery for congenital cystic adenomatoid malformation of the lung (CCAML) are not well documented, particularly regarding orthopaedic and respiratory follow-up (FU). The aim of this study was to assess the long-term pulmonary and orthopaedic outcomes of surgically treated CCAML in newborns, infants, and toddlers.

Materials and Methods Retrospective examination of prospectively recorded data of consecutive patients with CCAML who underwent surgery at our tertiary referral institution from January 2000 to December 2015 (newborns, infants, and toddlers). Clinical, radiological, and surgical data, as well as FU data were revised. A multidisciplinary team followed the patients after discharge at scheduled time points.

Results Seventy-seven patients were included. After surgery, patients were followed for a median of 8 years (range: 1–19 years) until they reached a median age of 8 years (range: 2–19 years). Thirty patients (39%) developed wheezing and 21 (27%) had lower respiratory tract infections (LRTIs) within 4 years of age. However, more than 50% of patients with respiratory symptoms underwent complete remission in the following 4 years. Thirty-one patients (40%) developed at least one minimal musculoskeletal deformity. Eighteen (23%) had scoliosis, 17 (22%) thoracic asymmetry, 10 (12%) pectus excavatum, and 5 (6%) winged scapula.

Conclusions Patients operated for CCAML had good overall outcomes despite pulmonary symptoms and musculoskeletal sequelae. Even though these issues are frequently paucisymptomatic, trying to use less-invasive procedures (such as minimally axillary open “muscle-sparing” thoracotomy or thoracoscopy) may reduce this burden. A structured multidisciplinary FU is required.

Keywords

congenital cystic adenomatoid malformation - lung malformations - follow-up - respiratory - orthopaedic - musculoskeletal


Publikationsverlauf

Eingereicht: 07. Mai 2023

Angenommen: 14. Juli 2023

Accepted Manuscript online:
17. Juli 2023

Artikel online veröffentlicht:
08. September 2023

© 2023. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Calzolari F, Braguglia A, Valfrè L, Dotta A, Bagolan P, Morini F. Outcome of infants operated on for congenital pulmonary malformations. Pediatr Pulmonol 2016; 51 (12) 1367-1372
    CrossrefPubMedSuche in Google Scholar
  • 2 Hall NJ, Stanton MP. Long-term outcomes of congenital lung malformations. Semin Pediatr Surg 2017; 26 (05) 311-316
    CrossrefPubMedSuche in Google Scholar
  • 3 Davenport M, Eber E. Long term respiratory outcomes of congenital thoracic malformations. Semin Fetal Neonatal Med 2012; 17 (02) 99-104
    CrossrefPubMedSuche in Google Scholar
  • 4 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
    CrossrefPubMedSuche in Google Scholar
  • 5 Zobel M, Gologorsky R, Lee H, Vu L. Congenital lung lesions. Semin Pediatr Surg 2019; 28 (04) 150821
    CrossrefPubMedSuche in Google Scholar
  • 6 van Leeuwen K, Teitelbaum DH, Hirschl RB. et al. Prenatal diagnosis of congenital cystic adenomatoid malformation and its postnatal presentation, surgical indications, and natural history. J Pediatr Surg 1999; 34 (05) 794-798 , discussion 798–799
    CrossrefPubMedSuche in Google Scholar
  • 7 Baird R, Puligandla PS, Laberge J-M. Congenital lung malformations: informing best practice. Semin Pediatr Surg 2014; 23 (05) 270-277
    CrossrefPubMedSuche in Google Scholar
  • 8 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
    CrossrefPubMedSuche in Google Scholar
  • 9 Akinkuotu AC, Olutoye OO. Congenital lung lesions. In: Mattei P, Nichol PF, Rollins II MD, Muratore CS. eds. Fundamentals of Pediatric Surgery. 2nd ed.. Cham: Springer International Publishing; 2017: 325-330
    Suche in Google Scholar
  • 10 Mon RA, Johnson KN, Ladino-Torres M. et al. Diagnostic accuracy of imaging studies in congenital lung malformations. Arch Dis Child Fetal Neonatal Ed 2019; 104 (04) F372-F377
    PubMedSuche in Google Scholar
  • 11 Cook J, Chitty LS, De Coppi P, Ashworth M, Wallis C. The natural history of prenatally diagnosed congenital cystic lung lesions: long-term follow-up of 119 cases. Arch Dis Child 2017; 102 (09) 798-803
    CrossrefPubMedSuche in Google Scholar
  • 12 David M, Lamas-Pinheiro R, Henriques-Coelho T. Prenatal and postnatal management of congenital pulmonary airway malformation. Neonatology 2016; 110 (02) 101-115
    CrossrefPubMedSuche in Google Scholar
  • 13 Laje P, Pearson EG, Simpao AF. et al. The first 100 infant thoracoscopic lobectomies: observations through the learning curve and comparison to open lobectomy. J Pediatr Surg 2015; 50 (11) 1811-1816
    CrossrefPubMedSuche in Google Scholar
  • 14 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 (01) 1-5
    Thieme ConnectPubMedSuche in Google Scholar
  • 15 Leblanc C, Baron M, Desselas E. et al. Congenital pulmonary airway malformations: state-of-the-art review for pediatrician's use. Eur J Pediatr 2017; 176 (12) 1559-1571
    CrossrefPubMedSuche in Google Scholar
  • 16 Ruchonnet-Metrailler I, Leroy-Terquem E, Stirnemann J. et al. Neonatal outcomes of prenatally diagnosed congenital pulmonary malformations. Pediatrics 2014; 133 (05) e1285-e1291
    CrossrefPubMedSuche in Google Scholar
  • 17 Lima M, D'Antonio S, Salvo ND. et al. Is it better to operate congenital lung malformations when patients are still asymptomatic?. J Indian Assoc Pediatr Surg 2021; 26 (03) 170-176
    CrossrefPubMedSuche in Google Scholar
  • 18 Thompson AJ, Sidebotham EL, Chetcuti PAJ, Crabbe DCG. Prenatally diagnosed congenital lung malformations-a long-term outcome study. Pediatr Pulmonol 2018; 53 (10) 1442-1446
    CrossrefPubMedSuche in Google Scholar
  • 19 Hijkoop A, van Schoonhoven MM, van Rosmalen J. et al. Lung function, exercise tolerance, and physical growth of children with congenital lung malformations at 8 years of age. Pediatr Pulmonol 2019; 54 (08) 1326-1334
    CrossrefPubMedSuche in Google Scholar
  • 20 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
    CrossrefPubMedSuche in Google Scholar
  • 21 Mattioli G, Pio L, Disma NM. et al. Congenital lung malformations: shifting from open to thoracoscopic surgery. Pediatr Neonatol 2016; 57 (06) 463-466
    CrossrefPubMedSuche in Google Scholar
  • 22 Conforti A, Aloi I, Trucchi A. et al. Asymptomatic congenital cystic adenomatoid malformation of the lung: is it time to operate?. J Thorac Cardiovasc Surg 2009; 138 (04) 826-830
    CrossrefPubMedSuche in Google Scholar
  • 23 Stocker JT. Cystic lung disease in infants and children. Fetal Pediatr Pathol 2009; 28 (04) 155-184
    CrossrefPubMedSuche in Google Scholar
  • 24 Dindo D, Demartines N, Clavien PA. Classification of surgical complications: a new proposal with evaluation in a cohort of 6336 patients and results of a survey. Ann Surg 2004; 240 (02) 205-213
    CrossrefPubMedSuche in Google Scholar
  • 25 Nuutinen S, Ronkainen E, Perhomaa M. et al. Long-term results of pediatric congenital pulmonary malformation: a population-based matched case-control study with a mean 7-year follow-up. Children (Basel) 2022; 10 (01) 71
    PubMedSuche in Google Scholar
  • 26 Dunn A, Pearce K, Callister R. et al. Exercise capacity is not decreased in children who have undergone lung resection early in life for congenital thoracic malformations compared to healthy age-matched children. Pediatr Pulmonol 2017; 52 (10) 1340-1348
    CrossrefPubMedSuche in Google Scholar
  • 27 Dincel A, Yilmaz Yegit C, Ergenekon AP. et al. Long-term respiratory outcomes of post-op congenital lung malformations. Pediatr Int 2021; 63 (06) 704-709
    CrossrefPubMedSuche in Google Scholar
  • 28 Lau C-T, Wong KKY. Long-term pulmonary function after lobectomy for congenital pulmonary airway malformation: is thoracoscopic approach really better than open?. J Pediatr Surg 2018; 53 (12) 2383-2385
    CrossrefPubMedSuche in Google Scholar
  • 29 Lau CT, Wong KKY, Tam P. Medium term pulmonary function test after thoracoscopic lobectomy for congenital pulmonary airway malformation: a comparative study with normal control. J Laparoendosc Adv Surg Tech A 2018; 28 (05) 595-598
    CrossrefPubMedSuche in Google Scholar
  • 30 Markel M, Derraugh G, Lacher M. et al. Musculoskeletal deformities in children with congenital thoracic malformations: a population-based cohort study. Pediatr Surg Int 2022; 38 (05) 731-736
    CrossrefPubMedSuche in Google Scholar
  • 31 Lam FKF, Lau C-T, Yu MO, Wong KKY. Comparison of thoracoscopy vs. thoracotomy on musculoskeletal outcomes of children with congenital pulmonary airway malformation (CPAM). J Pediatr Surg 2021; 56 (10) 1732-1736
    CrossrefPubMedSuche in Google Scholar
  • 32 Makita S, Kaneko K, Ono Y, Uchida H. Risk factors for thoracic and spinal deformities following lung resection in neonates, infants, and children. Surg Today 2017; 47 (07) 810-814
    CrossrefPubMedSuche in Google Scholar
  • 33 Faldini C, Manzetti M, Neri S. et al. Epigenetic and genetic factors related to curve progression in adolescent idiopathic scoliosis: a systematic scoping review of the current literature. Int J Mol Sci 2022; 23 (11) 5914
    CrossrefPubMedSuche in Google Scholar
  • 34 Johnson WR, Fedor D, Singhal S. Systematic review of surgical treatment techniques for adult and pediatric patients with pectus excavatum. J Cardiothorac Surg 2014; 9: 25
    CrossrefPubMedSuche in Google Scholar