Extralobar pulmonary sequestrations hiding congenital diaphragmatic defects: A case series

Elisa Negri; Noemi  Cantone; Elisa Severi; Gilda Belli; Francesca Tocchioni; Nicola Centonze; Enrico  Ciardini; Bruno  Noccioli

doi:10.47338/jns.v10.922

Authors

Elisa Negri Department of Pediatric Surgery, Meyer Children’s Hospital, Florence, Italy
Noemi Cantone Department of Neonatal Surgery, Meyer Children’s Hospital, Florence, Italy
Elisa Severi Department of Neonatal Surgery, Meyer Children’s Hospital, Florence, Italy
Gilda Belli Department of Neonatal and Intensive care Unit, Meyer Children’s Hospital, Florence, Italy.
Francesca Tocchioni Department of Neonatal Surgery, Meyer Children’s Hospital, Florence, Italy
Nicola Centonze Department of Neonatal Surgery, Meyer Children’s Hospital, Florence, Italy.
Enrico Ciardini Department of Pediatric Surgery, APSS Santa Chiara Hospital, Trento, Italy.
Bruno Noccioli Department of Neonatal Surgery, Meyer Children’s Hospital, Florence, Italy.

DOI:

https://doi.org/10.47338/jns.v10.922

Keywords:

Bronchopulmonary sequestration, Diaphragmatic hernia, Fetal MRI

Abstract

Background: Association between pulmonary sequestration and diaphragmatic hernia is well known. Extralobar sequestrations are masses of the non-functioning lung, surrounded by separate pleura, without bronchial communication, and with a systemic arterial blood supply. They may be placed in the thorax, within the diaphragm, or rarely in a sub-diaphragmatic position.

Case Series: We present three cases of extra-lobar extra-thoracic pulmonary sequestrations associated with different types of diaphragmatic defects. In none of the three cases, the diaphragmatic defect was detected prenatally.

Conclusion: Pulmonary sequestration may be involved in the embryological origin of the diaphragmatic defect. Simultaneously, it acts as an anatomical barrier and prevents the herniation of the abdominal content into the thorax. If extralobar pulmonary sequestration is diagnosed prenatally, a coexistent diaphragmatic hernia should always be considered.

Downloads

Download data is not yet available.

Metrics

Metrics Loading ...

References

Bratu I, Flageole H, Chen MF, Di Lorenzo M, Yazbeck S, Laberge JM. The multiple facets of pulmonary sequestration. J Pediatr Surg. 2001; 36:784-90.

Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung; a report of seven cases. J Pathol Bacteriol. 1946; 58:457-67.

Laje P, Martinez-Ferro M, Grisoni E, Dudgeon D. Intraabdominal pulmonary sequestration. A case series and review of the literature. J Pediatr Surg. 2006; 41:1309-12.

Kawamura N, Bhandal S. Coexistent congenital diaphragmatic hernia with extrapulmonary sequestration. Can Respir J. 2016;2016.

Savic B, Birtel F, Tholen W, Funke H, Knoche R. Lung sequestration : report of seven cases and review. Thorax. 1979; 34:96-101.

Grethel EJ, Farrell J, Ball RH. Does congenital diaphragmatic hernia associated with bronchopulmonary sequestration portend a better prognosis? Fetal Diagn Ther. 2008; 23:250-3.

Cruz SM, Akinkuotu AC, Cass DL, Lee TC, Cassady CI, Mehollin-Ray AR, et al. Space occupying lesions in the presence of congenital diaphragmatic hernia. J Pediatr Surg. 2016; 51:710-13.

Soni S, Moldenhauer JS, Rintoul N, Adzick NS, Hedrick HL, Khalek N. Perinatal outcomes in fetuses prenatally diagnosed with a congenital diaphragmatic hernia and concomitant lung lesions: A 10-year review. Fetal Diagn Ther. 2020; 47:630-35.

Gerle RD, Jaretzki A, Ashley CA, Berne S. Congenital bronchopulmonary-foregut malformation. N Engl J Med. 1968; 278:1413-9.

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

Harris K. Extralobar sequestration with congenital diaphragmatic hernia: a complicated case study. Neonatal Netw. 2004; 23:7-24.

Luet'Ic T, Crombleholme TM, Semple JP, D'Alton M. Early prenatal diagnosis of bronchopulmonary sequestration with associated diaphragmatic hernia. J Ultrasound Med. 1995; 14:533-5.

Kim HM, Hwang JH, Kim MJ, Cha HH, Seong WJ. Postnatally diagnosed coexisting congenital diaphragmatic hernia with pulmonary sequestration: A report of two cases. Obstet Gynecol Sci. 2020; 63:529-33.

Levine D. Fetal magnetic resonance imaging. J Matern Neonatal Med. 2004;v15:85-94.

Xu G, Zhou J, Zeng S, Zhang M, Ouyang Z, Zhao Y, et al. Prenatal diagnosis of fetal intraabdominal extralobar pulmonary sequestration: a 12-year 3-center experience in China. Sci Rep. 2019; 9:1-9.

Son SA, Do YW, Kim YE, Lee SM, Lee DH. Infarction of torsed extralobar pulmonary sequestration in adolescence. Gen Thorac Cardiovasc Surg. 2020; 68:77-80.