Tratamento cirúrgico das malformações broncopulmonares em crianças: experiência em um centro Brasileiro
Surgical treatment of bronchopulmonary malformations in children: experience in a Brazilian center
Átila Magalhães Victótia; Fabio Botelho; Clecio Picarro; Paulo Custódio Furtado Cruzeiro; Sherif Emil; José Carlos Fraga; Marcelo Eller Miranda
Resumo
Introdução: As malformações broncopulmonares (MBP) são anomalias do trato respiratório inferior que incluem malformações congênitas das vias aéreas pulmonares (MCVAP), cistos broncogênicos (CB), sequestros broncopulmonares (SBP) e enfisema lobar congênito (ELC). A detecção pré-natal em países de baixa e média renda é menos comum do que em países de alta renda. Este estudo pretende mostrar a experiência na abordagem cirúrgica da MBP em um centro brasileiro, com ênfase na evolução clínica e nos resultados cirúrgicos, conforme a época do diagnostico (pré-natal versus pós-natal).
Métodos: Uma análise retrospectiva dos prontuários médicos foi realizada para pacientes com menos de 18 anos que foram submetidos à cirurgia para MBP em um centro de referência de um país de renda média entre 2000 e 2021. Com base no momento do diagnóstico da MBP, as crianças foram divididas em dois grupos: pré-natal e pós-natal. Esses grupos foram avaliados quanto à idade na cirurgia, histórico de pneumonia antes da operação, resultados cirúrgicos (complicações peri- e pós-operatórias, duração da ventilação mecânica, duração do dreno torácico, tempo de internação) e o tipo histológico de MBP.
Resultados: Na coorte de 66 pacientes, 43 (65,1%) tinham diagnóstico pré-natal de MBP, enquanto 23 (34,8%) foram identificados após o nascimento. Em comparação com os pacientes diagnosticados no pré-natal, aqueles diagnosticados após o nascimento foram submetidos à cirurgia com idade maior (média de 978 dias ± 1341,0 versus média de 200 dias ± 360,9; p<0,01), e apresentaram maior incidência de pneumonia antes da cirurgia (65% vs. 12%, p < 0,01). Não houve associação entre o momento do diagnóstico de MBP e os resultados pós-cirúrgicos. Todos os pacientes com SBP estavam no grupo pré-natal, e todos os pacientes com ELC estavam no grupo pós-natal. Houve maior prevalência de MCVAP no grupo pré-natal em comparação com o grupo pós-natal (72% vs. 39%, p < 0,01).
Conclusão: Em um centro brasileiro, cerca de 2/3 dos pacientes tiveram o diagnóstico intrauterino das malformações broncopulmonares e foram atendidos precocemente em centro de neonatologia. Os pacientes com diagnóstico das MBP somente após o nascimento tinham maior probabilidade de apresentar pneumonia e passar por cirurgia com idade maior do que os pacientes com diagnóstico intrauterino. Estudos prospectivos e multicêntricos, incluindo pacientes assintomáticos tratados conservadoramente, sem intervenções cirúrgicas, e os pacientes operados por videotoracoscopia, seriam bem indicados para se avaliar futuramente a evolução das crianças com MBP, e estabelecer protocolos adequados à realidade brasileira.
Palavras-chave
Abstract
Introduction: Bronchopulmonary malformations (BPM) are lower respiratory tract anomalies that include congenital malformations of the pulmonary airways (CMPA), bronchogenic cysts (BC), bronchopulmonary sequestrations (BPS), and congenital lobar emphysema (CLE). Prenatal detection in low- and middle-income countries is less common than in high-income ones. This study aims to show the experience in the surgical approach to BPM in a Brazilian center, with emphasis on clinical evolution and surgical results, according to the time of diagnosis (prenatal versus postnatal).
Methods: We retrospectively analyzed medical records of patients under the age of 18 who underwent surgery for BPM at a referral center in a middle-income country between 2000 and 2021. Based on the time of BPM diagnosis, we divided the children into two groups: prenatal and postnatal. These groups were evaluated in terms of age at surgery, history of pneumonia before the operation, surgical outcomes (perioperative and postoperative complications, duration of mechanical ventilation, duration of chest tube, length of hospital stay), and histological type of BPM.
Results: In the cohort of 66 patients, 43 (65.1%) had a prenatal diagnosis of BPM, while 23 (34.8%) were identified after birth. Compared with patients diagnosed prenatally, those diagnosed after birth underwent surgery at a higher age (mean of 978 days ± 1341.0 versus mean of 200 days ± 360.9; p<0.01), and had a higher incidence of pneumonia before surgery (65% vs. 12%, p < 0.01). There was no association between the time of BPM diagnosis and postoperative outcomes. All patients with BPS were in the prenatal group, and all patients with CLE were in the postnatal one. There was a higher prevalence of CMPA in the prenatal group compared with the postnatal one (72% vs. 39%, p < 0.01).
Conclusion: In a Brazilian center, approximately 2/3 of the patients had an intrauterine diagnosis of bronchopulmonary malformations and were treated early at a neonatology center. Patients diagnosed with BPM only after birth were more likely to have pneumonia and undergo surgery at an older age than patients with an intrauterine diagnosis. Prospective, multicenter studies, including asymptomatic patients treated conservatively, without surgical interventions, and patients operated by video-assisted thoracoscopy, would be well indicated to evaluate the future evolution of children with BPM and to establish protocols appropriate to the Brazilian reality.
Keywords
Referências
1 Ferreira HP, Fischer GB, Felicetti JC, Camargo J, Andrade CF. Surgical treatment of congenital lung malformations in pediatric patients. J Bras Pneumol. 2010;36(2):175-80. doi: 10.1590/s1806-37132010000200004.
2 Hermelijn SM, Zwartjes RR, Tiddens HAWM, Cochius-den Otter SCM, Reiss IKM, Wijnen RMH, et al. Associated Anomalies in Congenital Lung Abnormalities: A 20-Year Experience. Neonatology. 2020;117(6):697-703. doi: 10.1159/000509426.
3 Fowler DJ, Gould SJ. The pathology of congenital lung lesions. Semin Pediatr Surg. 2015;24(4):176-82. doi: 10.1053/j.sempedsurg.2015.02.002.
4 Wong KKY, Flake AW, Tibboel D, Rottier RJ, Tam PKH. Congenital pulmonary airway malformation: advances and controversies. Lancet Child Adolesc Health. 2018;2(4):290-7. doi: 10.1016/S2352-4642(18)30035-X.
5 Gonçalves LF, Lee W, Mody S, Shetty A, Sangi-Haghpeykar H, Romero R. Diagnostic accuracy of ultrasonography and magnetic resonance imaging for the detection of fetal anomalies: a blinded case-control study. Ultrasound Obstet Gynecol. 2016;48(2):185-92. doi: 10.1002/uog.15774.
6 Mon RA, Johnson KN, Ladino-Torres M, Heider A, Mychaliska GB, Treadwell MC, et al. Diagnostic accuracy of imaging studies in congenital lung malformations. Arch Dis Child Fetal Neonatal Ed. 2019;104(4):F372-F377. doi: 10.1136/archdischild-2018-314979.
7 Kunisaki SM. Narrative review of congenital lung lesions. Transl Pediatr. 2021;10(5):1418-31. doi: 10.21037/tp-20-133.
8 Adams S, Jobson M, Sangnawakij P, Heetun A, Thaventhiran A, Johal N, et al. Does thoracoscopy have advantages over open surgery for asymptomatic congenital lung malformations? An analysis of 1626 resections. J Pediatr Surg. 2017;52(2):247-51. doi: 10.1016/j.jpedsurg.2016.11.014.
9 Salles M, Deschildre A, Bonnel C, Dubos JP, Bonnevalle M, Devismes L, et al. Diagnosis and treatment of congenital bronchopulmonary malformations. A review of 32 cases. Arch Pediatr. 2005;12(12):1703-8. doi: 10.1016/j.arcped.2005.09.013.
10 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. Ann Surg. 2004;240(2):205-13. doi: 10.1097/01.sla.0000133083.54934.ae.
11 Adams NC, Victoria T, Oliver ER, Moldenhauer JS, Adzick NS, Colleran GC. Fetal ultrasound and magnetic resonance imaging: a primer on how to interpret prenatal lung lesions. Pediatr Radiol. 2020;50(13):1839-54. doi: 10.1007/s00247-020-04806-x.
12 Botelho F, Viana RFR, Emil SGS, Puligandla PS, Piçarro C, Cruzeiro PCF, et al. Gastroschisis prognostic score successfully identifies Brazilian newborns with high-risk gastroschisis. J Pediatr Surg. 2022;57(10):298-302. doi: 10.1016/j.jpedsurg.2022.02.009.
13 Stanton M, Njere I, Ade-Ajayi N, Patel S, Davenport M. Systematic review and meta-analysis of the postnatal management of congenital cystic lung lesions. J Pediatr Surg. 2009;44:1027-33.
14 de Doenças S de E da SC de C. Informação de vigilância das Pneumonias e Meningites bacterianas. Instituto Adolfo Lutz; 2021.
15 Sullivan KJ, Li M, Haworth S, Chernetsova E, Wayne C, Kapralik J, et al. Optimal age for elective surgery of asymptomatic congenital pulmonary airway malformation: a meta-analysis. Pediatr Surg Int. 2017;33(6):665-75. doi: 10.1007/s00383-017-4079-2.
16 Kapralik J, Wayne C, Chan E, Nasr A. Surgical versus conservative management of congenital pulmonary airway malformation in children: A systematic review and meta-analysis. J Pediatr Surg. 2016;51(3):508-12. doi: 10.1016/j.jpedsurg.2015.11.022.
17 Downard CD, Calkins CM, Williams RF, Renaud EJ, Jancelewicz T, Grabowski J, et al. Treatment of congenital pulmonary airway malformations: a systematic review from the APSA outcomes and evidence based practice committee. Pediatr Surg Int. 2017;33(9):939-53. doi: 10.1007/s00383-017-4098-z.
18 Singh R, Davenport M. The argument for operative approach to asymptomatic lung lesions. Semin Pediatr Surg. 2015;24(4):187-95. doi: 10.1053/j.sempedsurg.2015.02.003.
19 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(9):798-803. doi: 10.1136/archdischild-2016-311233.
20 Hall NJ, Chiu PPL, Langer JC. Morbidity after elective resection of prenatally diagnosed asymptomatic congenital pulmonary airway malformations. Pediatr Pulmonol. 2016;51(5):525-30. doi: 10.1002/ppul.23255.
21 Naito Y, Beres A, Lapidus-Krol E, Ratjen F, Langer JC. Does earlier lobectomy result in better long-term pulmonary function in children with congenital lung anomalies? A prospective study. J Pediatr Surg. 2012;47(5):852-6. doi: 10.1016/j.jpedsurg.2012.01.037.
22 Style CC, Cass DL, Verla MA, Cruz SM, Lau PE, Lee TC, et al. Early vs late resection of asymptomatic congenital lung malformations. J Pediatr Surg. 2019;54(1):70-74. doi: 10.1016/j.jpedsurg.2018.10.035.
23 Safa N, Wei S, Saran N, Guadagno E, Laberge JM, Emil S. Musculoskeletal deformities after thoracic surgery in children: An observational long-term follow-up study. J Pediatr Surg. 2021;56(1):136-141. doi:10.1016/j.jpedsurg.2020.09.024
24 Shah AA, Shakoor A, Zogg CK, Oyetunji T, Ashfaq A, Garvey EM, et al. Influence of sub-specialty surgical care on outcomes for pediatric emergency general surgery patients in a low-middle income country. Int J Surg. 2016;29:12-8. doi: 10.1016/j.ijsu.2016.03.007.
Submetido em:
09/08/2024
Aceito em:
10/12/2024