Congenital Lobar Emphysema: Perioperative Evaluation and Management
DOI:
https://doi.org/10.1532/hsf.3823Keywords:
Congenital lobar overinflation, Lobectomy, Neonatal respiratory distressAbstract
Background: Congenital lobar emphysema (CLE) is a lung malformation characterized by overdistension and air trapping in the affected lobe. It is one of the causes of neonatal and infantile respiratory distress. This study aimed to evaluate our experience regarding perioperative and surgical management in children with CLE.
Methods: A retrospective observational study was done for all CLE patients who underwent surgery at Menoufia University Hospital. Perioperative data collected included demographic, clinical, and radiological findings, as well as operative and postoperative data.
Results: We included 30 neonates and infants who suffered from CLE between January 2013 and December 2020; the mean age was 111.43 ± 65.19 days, and 21 were males. All cases presented with respiratory distress; 19 had cyanosis, and 15 had recurrent pneumonia and fever. Plain chest x-ray and computed tomography (CT) revealed emphysema in all cases. Lobectomy was done in all cases; the mean age at surgery was 147.58 ± 81.49 days. Postoperative complications occurred in 5 patients, and 2 of them needed mechanical ventilation. The follow-up duration ranged from 3 months to 1 year (except 1 case lost to follow-up after 3 months), and all patients were doing well.
Conclusion: CLE is a rare bronchopulmonary malformation that requires a high index of clinical suspicion, especially in persistent and recurrent infantile respiratory distress. Chest CT is the most useful diagnostic modality. Early management of CLE improves outcome and prevents life-threatening complications. Surgical management is the treatment of choice in our center, without recorded mortality.
References
Aslan AT, Yalcin E, Ozcelik, et al. Foreign-body aspiration mimicking congenital lobar emphysema in a forty-eight-day-old girl. Pediatr Pulmonol 2005;39:189-191.
Berrocal T, Madrid C, Novo S, et al. Congenital anomalies of the tracheobronchial tree, lung, and mediastinum: Embryology, radiology, and pathology. Radiographics 2004;24:e17.
Bush A, Harcout J, Hewitt RJ, Nicholson AG. Congenital lung disease. In: Wilmott RW, Deterding R, Li A, et al., editors. Kendig’s Disorders of the Respiratory Tract in Children. 9th ed. Philadelphia: Elsevier; 2019;321-322.
Cataneo DC, Rodrigues OR, Hasimoto EN, et al. Congenital lobar emphysema: 30-year case series in two university hospitals. J Bras Pneumol 2013;39:418-426.
Ceran S, Altuntas B, Sunam GS, et al. Congenital lobar emphysema: Is surgery routinely necessary? Afr J Paediatr Surg 2010;7:36-37.
Correia-Pinto J, Gonzaga S, et al. Congenital lung lesions underlying molecular mechanisms. Semin Pediatr Surg 2010;19:171-179.
Davis C, Lawrence J, Kenneth C, et al. Congenital lobar emphysema. South Med J 1979;72:696-698.
Demir OF, Hangul M, Kose M. Congenital lobar emphysema: Diagnosis and treatment options. Int J Chron Obstruct Pulmon Dis 2019;14:921-928.
Fierro JL, Hysinger EB, Piccione J, et al. The role of selective balloon occlusion in preoperative planning for infant pulmonary lobar hyperinflflation. Pediatr Allergy Immunol Pulmonol 2018;31:186-190.
Karnak I, Senocak ME, Ciftci AO, et al. Congenital lobar emphysema: Diagnostic and therapeutic considerations. J Pediatr Surg 1999;34:1347-1351.
Kerstine KH, Van Natta TL, Burkhart HM, et al. Congenital lung diseases. In: Sellke FW, Del Nido PJ, Swanson SJ, editors. Sabiston & Spencer Surgery of the Chest. Vol. 1, 8th Ed. Philadelphia: Saunders Elsevier; 2010;129-150.
Khemiri M, Ouederni M, Ben Mansour F, et al. Bronchogenic cyst: An uncommon cause of congenital lobar emphysema. Respir Med 2008;102:1663-1666.
Kumar B, Agrawal LD, Sharma SB. Congenital bronchopulmonary malformations: A single-centre experience and a review of literature. Ann Thorac Med 2008;3:135-139.
Kunisaki SM, Saito JM, Fallat ME, et al. Current operative management of congenital lobar emphysema in children: A report from the Midwest Pediatric Surgery Consortium. J Pediatr Surg 2019;54:1138-1142.
Lincoln JC, Stark J, Subramanian S, et al. Congenital lobar emphysema. Ann Surg 1971;173:55-62.
Man DW, Hamdy MH, Hendry GM, et al. Congenital lobar emphysema: Problems in diagnosis and management. Arch Dis Child 1983;58:709-712.
Markowitz RI, Mercurio MR, Vahjen GA, et al. Congenital lobar emphysema. The roles of CT and V/Q scan. Clin Pediatr (Phila) 1989;28:19-23.
Mei-Zahav M, Konen O, Manson D, et al. Is congenital lobar emphysema a surgical disease? J Pediatr Surg 2006;41:1058-1061.
Mendeloff EN. Sequestrations, congenital cystic adenomatoid malformations, and congenital lobar emphysema. Semin Thorac Cardiovasc Surg 2004;16:209-214.
Michelson E. Clinical spectrum of infantile lobar emphysema. Ann Thorac Surg 1977;24:182-196.
Nazem M, Hosseinpour M. Evaluation of early and late complications in patients with congenital lobar emphysema: A 12 year experience. Afr J Paediatr Surg 2010;7:144-146.
Olutoye OO, Coleman BG, Hubbard AM, et al. Prenatal diagnosis and management of congenital lobar emphysema. J Pediatr Surg 2000;35:792-795.
Ozcelik U, Göçmen A, Kiper N, et al. Congenital lobar emphysema: Evaluation and long-term follow up of thirty cases at a single centre. Pediatr Pulmonol 2003;35:384-391.
Paramalingam S, Parkinson E, Sellars M, et al. Congenital segmental emphysema: ;n evolving lesion. Eur J Pediatr Surg 2010;20:e78-e81.
Perea L, Blinman T, Piccione J, et al. Bilateral congenital lobar emphysema: Staged management. J Pediatr Surg 2017;52:1442-1445.
Rahman N, Lakhoo K. Comparison between open and thoracoscopic resection of congenital lung lesions. J Pediatr Surg 2009;44:333-336.
Thacker PG, Rao AG, Hill JG, et al. Congenital lung anomalies in children and adults current concepts and imaging findings. Radiol Clin N Am 2014;52:155-181.
Thakral CL, Maji DC, Sajwani MJ. Congenital lobar emphysema: Experience with 21 cases. Pediatr Surg Int 2001;17:88-91.
Ulku R, Onat S, Ozçelik C. Congenital lobar emphysema: Differential diagnosis and therapeutic approach. Pediatr Int 2008;50:658-661.
Zoeller C, Ure BM, Dingemann J. Perioperative complications of video-assisted thoracoscopic pulmonary procedures in neonates and infants. Eur J Pediatr Surg 2018;28:163-70.
