ABSTRACT

Introduction: Prenatal ultrasonography currently represents presentation form of bronchopulmonary malformations. To 90% manifest in neonates with obvious radiological abnormalities and symptomatic progression where surgical resection is clearly indicated. In ‹ 4% causing mass effect with mediastinal shift, hydrops and fetal death and other extreme behave «asymptomatic» with apparent involution and its management has been controversial: early elective resection or radiologic expectant management to document the regression or development complications. It analyzes the literature about the controversy: operate or not to operate. Description of cases: Three fetuses with prenatal diagnosis of intrathoracic solid mass, asymptomatic newborns: one with hydrothorax and other without apparent prenatal involution both with abnormal irrigation compatible with extralobar sequestration, the third with a paracardiac mass where imaging studies did not diagnostic a extralobar sequestration of ectopic location and synchronous with cystic adenomatoid malformation, only transoperative and pathological findings confirmed the diagnosis. Discussion: In asymptomatic patients all bronchopulmonary malformations could be diagnosed and surgically resected for unrecognized their natural history and the low rate of natural regression, radiological proposals are not effective for monitoring.

KEYWORDS

Extralobar sequestration, elective resection, asymptomatic malformation.

REFERENCES

1. 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(5):1027-1033. doi: 10.1016/j.jpedsurg.2008.10.118.

2. Parikh D, Samuel M. Congenital cystic lung lesions: is surgical resection essential? Pediatr Pulmonol 2005;40(6):533-537.

3. Laberge JM, Puligandla P, Flageole H. Asymptomatic congenital lung malformations. Semin Pediatr Surg 2005;1481):16-33.

4. Laberge JM, Bratu I, Flageole H. The management of asymptomatic congenital lung malformations. Paediatr Respir Rev 2004;5 Suppl A:S305-S312.

5. Adzick NS. Management of fetal lung lesions. Clin Perinatol 2009;36(2):363-376. x. doi: 10.1016/j.clp.2009.03.001.

6. Aziz D, Langer JC, Tuuha SE, Ryan G, Ein SH, Kim PC. Perinatally diagnosed asymptomatic congenital cystic adenomatoid malformation: to resect or not? J Pediatr Surg 2004;39(3):329-334.

7. Huang EY, Monforte HL, Shaul DB. Extralobar pulmonary sequestration presenting with torsion. Pediatr Surg Int 2004;20(3):218-220.

8. Rubin EM, Garcia H, Horowitz MD, Guerra JJ Jr. Fatal massive hemoptysis secondary to intralobar sequestration. Chest 1994;106(3):954-955.

9. Avishai V, Dolev E, Weissberg D, Zajdel L, Priel IE. Extralobar sequestration presenting as massive hemothorax. Chest 1996;109(3):843-845.

10. Hancock BJ, Di Lorenzo M, Youssef S, Yazbeck S, Marcotte JE, Collin PP. Childhood primary pulmonary neoplasms. J Pediatr Surg 1993;28(9):1133-1136.

11. Al-Bassam A, Al-Rabeeah A, Al-Nassar S, et al. Congenital cystic disease of the lung in infants and children (experience with 57 cases). Eur J Pediatr Surg 1999;9(6):364-368.

12. Tsai AY, Liechty KW, Hedrick HL, et al. Outcomes after postnatal resection of prenatally diagnosed asymptomatic cystic lung lesions. J Pediatr Surg 2008;43(3): 513-517. doi: 10.1016/j.jpedsurg.2007.10.032.

13. Cass DL, Crombleholme TM, Howell LJ, Stafford PW, Ruchelli ED, Adzick NS. Cystic lung lesions with systemic arterial blood supply: a hybrid of congenital cystic adenomatoid malformation and bronchopulmonary sequestration. J Pediatr Surg 1997;32(7):986-990.

14. Chen HW, Hsu WM, Lu FL, et al. Management of congenital cystic adenomatoid malformation and bronchopulmonary sequestration in newborns. Pediatr Neonatol 2010;51(3):172-177. doi: 10.1016/S1875-9572(10)60032-0.

15. Yildiz K, Ozcan N, Cebi M, Köse N, Karakaya F. Intrapericardial extralobar pulmonary sequestration: unusual cause of hydrops fetalis. J Ultrasound Med 2005;24(3):391-393.

16. Benatar A, Vaughan J, Nicolini U, Trotter S, Corrin B, Lincoln C. Prenatal pericardiocentesis: its role in the management of intrapericardial teratoma. Obstet Gynecol 1992;79(5 (Pt 2)):856-859.

17. Todros T, Gaglioti P, Presbitero P. Management of a fetus with intrapericardial teratoma diagnosed in utero. J Ultrasound Med 1991;10(5):287-290.

18. Sydorak RM, Kelly T, Feldstein VA, et al. Prenatal resection of a fetal pericardial teratoma. Fetal Diagn Ther 2002;17(5):281-285.

19. Cho MJ, Kim DY, Kim SC, Kim KS, Kim EA, Lee BS. Embolization versus surgical resection of pulmonary sequestration: clinical experiences with a thoracoscopic approach. J Pediatr Surg 2012;47:2228-2233. doi: 10.1016/j.jpedsurg.2012.09.013.

20. Yeh CN, Wang JN, Tsai YC, Yao CT, Lin CS, Wu JM. Coil embolization of pulmonary sequestration in two infants: a safe alternative management. Acta Paediatr Taiwan 2006;47(2):88-91.

21. Lee KH, Sung KB, Yoon HK, et al. Transcatheter arterial embolization of pulmonary sequestration in neonates: long term follow up results. J Vasc Interv Radiol 2003;14(3):363-367.

22. Brown EG, Marr C, Farmer DL. Extralobar pulmonary sequestration: The importance of intraoperative vigilance. J Ped Surg Case Reports 2013;1(4):74-76. doi:10.1016/j.epsc.2013.02.015

23. Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration: report of seven cases and review of 540 published cases. Thorax 1979;34(1):96-101.

24. Osaki T, Kodate M, Takagishi T, Nomi M, Murakami J, Yamamoto H. Unique extralobar sequestration with atypical location and aberrant vessels. Ann Thorac Surg 2010:90(5);1711-1712. doi: 10.1016/j.athoracsur.2010.04.031.

25. Samuel M, Burge DM. Management of antenatally diagnosed pulmonary sequestration associated with congenital cystic adenomatoid malformation. Thorax 1999;54(8):701-706.

26. Di Prima FA, Bellia A, Inclimona G, Grasso F, Teresa M, Cassaro MN. Antenatally diagnosed congenital cystic adenomatoid malformations (CCAM): Research review. J Prenat Med 2012;6(2):22-30.

27. Singh R, Davenport M. The argument for operative approach to asymptomatic lung lesions. Semin Pediatr Surg 2015;24(4):187-195. doi: 10.1053/j.sempedsurg.2015.02.003.