Congenital lobar hyperinflation (CHL): A case report with early and differential diagnosis in Neonatal Respiratory Syndrome
DOI:
https://doi.org/10.33448/rsd-v14i6.49132Keywords:
Congenital malformation, Respiratory distress, COVID.Abstract
Congenital lobar hyperinflation (CHL) is a rare lung malformation with a prevalence of 1 in 20,000 to 30,000 births. This is a descriptive study of a clinical case based on data collection during patient follow-up, review of medical records and photographic records. This study aims to elucidate several aspects of CHL, from its diagnosis to prognosis, as a differential diagnosis in Neonatal Respiratory Syndrome. The study describes a clinical case of a newborn who presented respiratory distress in the first hours of life and was diagnosed with CHL. This study highlights the importance of early diagnosis of CHL, where neonatal respiratory failure can be confused with viral infection. Knowledge of this malformation is essential to avoid complications and ensure adequate treatment.
References
Ankermann, T., Oppermann, H. C., Engler, S., Leuschner, I., & Von Kaisenberg, C. S. (2004). Congenital masses of the lung, cystic adenomatoid malformation versus congenital lobar emphysema: prenatal diagnosis and implications for postnatal treatment. Journal of ultrasound in medicine, 23(10), 1379-1384.
Ayed, A. K., & Owayed, A. (2003). Pulmonary resection in infants for congenital pulmonary malformation. Chest, 124(1), 98-101.
BH, L. (1979). Congenital malformations and genetic disorders of the respiratory tract (larynx, trachea, bronchi and lungs). Am Rev Respir Dis, 120, 151-185.
Cataneo, D. C., Rodrigues, O. R., Hasimoto, E. N., Schmidt Jr, A. F., & Cataneo, A. J. M. (2013). Congenital lobar emphysema: 30-year case series in two university hospitals. Jornal Brasileiro de Pneumologia, 39, 418-426.
DeLuca, F. G., & Wesselhoeft, C. W. (1978). Surgically treatable causes of neonatal respiratory distress. Clinics in Perinatology, 5(2), 377-394.
Dillman, J. R., Sanchez, R., Ladino-Torres, M. F., Yarram, S. G., Strouse, P. J., & Lucaya, J. (2011). Expanding upon the unilateral hyperlucent hemithorax in children. Radiographics, 31(3), 723-741.
Duan, M., Wang, L., Cao, Y., Li, Z., Yang, W., Rao, X., & Xiao, K. (2005). Results of surgical treatment of congenital cystic lung disease. The Thoracic and Cardiovascular Surgeon, 53(1), 61-64.
Eber, E. (2007, June). Antenatal diagnosis of congenital thoracic malformations: early surgery, late surgery, or no surgery?. In Seminars in respiratory and critical care medicine, 28(3), 355-366. Copyright© 2007 by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA.
Fowler, D. J., & Gould, S. J. (2015, August). The pathology of congenital lung lesions. In Seminars in pediatric surgery, 24(4), 176-182. WB Saunders.
Hernanz-Schulman, M. (1993). Cysts and cystlike lesions of the lung. Radiologic Clinics of North America, 31(3), 631-649.
Kaptanoğlu, M., Yüksel, M., & Kaptanoğlu, M. (2004). Konjenital akciğer hastalıları. Pediatrik Göğüs Cerrahisi, 1, 179-184.
Khosa, J. K., Leong, S. L., & Borzi, P. A. (2004). Congenital cystic adenomatoid malformation of the lung: indications and timing of surgery. Pediatric surgery international, 20, 505-508.
Kravitz, R. M. (1994). Congenital malformations of the lung. Pediatric Clinics of North America, 41(3), 453-472.
Kunisaki, S. M., Saito, J. M., Fallat, M. E., Peter, S. D. S., Kim, A. G., Johnson, K. N., ... & Midwest Pediatric Surgery Consortium. (2019). Current operative management of congenital lobar emphysema in children: a report from the Midwest Pediatric Surgery Consortium. Journal of pediatric surgery, 54(6), 1138-1142.
Markowitz, R. I., Mercurio, M. R., Vahjen, G. A., Gross, I., & Touloukian, R. J. (1989). Congenital lobar emphysema: the roles of CT and V/Q scan. Clinical pediatrics, 28(1), 19-23.
Mendeloff, E. N. (2004, September). Sequestrations, congenital cystic adenomatoid malformations, and congenital lobar emphysema. In Seminars in Thoracic and Cardiovascular Surgery, 16(3), 209-214. WB Saunders.
Nuchtern, J. G., & Harberg, F. J. (1994, November). Congenital lung cysts. In Seminars in Pediatric surgery, 3(4), 233-243.
Olutoye, O. O., Coleman, B. G., Hubbard, A. M., & Adzick, N. S. (2000). Prenatal diagnosis and management of congenital lobar emphysema. Journal of pediatric surgery, 35(5), 792-795.
Pardes, J. G., Auh, Y. H., Blomquist, K., Kazam, E., & Magid, M. (1983). CT diagnosis of congenital lobar emphysema. Journal of computer assisted tomography, 7(6), 1095-1097.
Pereira A. S. et al. (2018). Metodologia da pesquisa científica. [free e-book]. Editora UAB/NTE/UFSM.
Raymond, S. L., Sacks, M. A., Hashmi, A., Robertson, J. O., Moores, D., Tagge, E. P., ... & Khan, F. A. (2023). Short-term outcomes of thoracoscopic versus open lobectomy for congenital lung malformations. Pediatric Surgery International, 39(1), 155.
Roy Choudhury, S., Chadha, R., Mishra, A., Kumar, V., Singh, V., & Dubey, N. K. (2007). Lung resections in children for congenital and acquired lesions. Pediatric surgery international, 23, 851-859.
Rusakow, L. S., & Khare, S. (2001). Radiographically occult congenital lobar emphysema presenting as unexplained neonatal tachypnea. Pediatric pulmonology, 32(3), 246-249.
Schwartz, M. Z., & Ramachandran, P. (1997). Congenital malformations of the lung and mediastinum—a quarter century of experience from a single institution. Journal of pediatric surgery, 32(1), 44-47.
Shanmugam, G., MacArthur, K., & Pollock, J. C. (2005). Congenital lung malformations—antenatal and postnatal evaluation and management. European Journal of Cardio-thoracic Surgery, 27(1), 45-52.
Shanti, C. M., & Klein, M. D. (2008, February). Cystic lung disease. In Seminars in pediatric surgery, 17(1), 2-8. WB Saunders.
Singh, R., & Davenport, M. (2015, August). The argument for operative approach to asymptomatic lung lesions. In Seminars in pediatric surgery, 24(4), 187-195. WB Saunders.
Stanton, M. (2015, August). The argument for a non-operative approach to asymptomatic lung lesions. In Seminars in pediatric surgery, 24(4), 183-186. WB Saunders.
Stigers, K. B., Woodring, J. H., & Kanga, J. F. (1992). The clinical and imaging spectrum of findings in patients with congenital lobar emphysema. Pediatric pulmonology, 14(3), 160-170.
Stocker, J. T., Drake, R. M., & Madewell, J. E. (1978). Cystic and congenital lung disease in the newborn. Perspectives in pediatric pathology, 4, 93-154.
Tander, B., Yalçın, M., Yılmaz, B., Karadağ, C. A., & Bulut, M. (2003). Congenital lobar emphysema: a clinicopathologic evaluation of 14 cases. European journal of pediatric surgery, 13(02), 108-111.
Thakral, C. L., Maji, D. C., & Sajwani, M. J. (2001). Congenital lobar emphysema: experience with 21 cases. Pediatric surgery international, 17, 88-91.
Toassi, R. F. C. & Petry, P. C. (2021; Yin, 2015). Metodologia científica aplicada à área da Saúde. (2ed). Editora da UFRGS.
Truitt, A. K., Carr, S. R., Cassese, J., Kurkchubasche, A. G., Tracy Jr, T. F., & Luks, F. I. (2006). Perinatal management of congenital cystic lung lesions in the age of minimally invasive surgery. Journal of pediatric surgery, 41(5), 893-896.
Wasilewska, E., Lee, E. Y., & Eisenberg, R. L. (2012). Unilateral hyperlucent lung in children. American Journal of Roentgenology, 198(5), W400-W414.
Wu, Y., Xi, L., Pan, Z., Wu, C., & Li, H. (2023). Torsed extralobar pulmonary sequestration associated with congenital lobar emphysema: a rare entity. Therapeutic Advances in Respiratory Disease, 17, 17534666231164535.
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