Full Text (PDF)
Case Report

Hybrid Lesion: Extrapulmonary Sequestration with Cystic Adenomatoid Malformation: A Rare Case Report

Sapna Patel, Pallavi BR, Sheela Devi CS, Devi Sreelakshmi

Author Information

Licence:

Attribution-Non-commercial 4.0 International (CC BY-NC 4.0)

This license enables reusers to distribute, remix, adapt, and build upon the material in any medium or format for noncommercial purposes only, and only so long as attribution is given to the creator.


Indian Journal of Forensic Medicine and Pathology 17(3):p 207-210, July- September 2024. | DOI: https://doi.org/10.21088/ijfmp.0974.3383.17324.9

How Cite This Article:

Patel S, Pallavi BR, Devi SCS, et al. Hybrid Lesion: Extrapulmonary Sequestration with Cystic Adenomatoid Malformation: A Rare Case Report. Indian J Forensic Med Pathol. 2024;17(3):207-210.

Timeline

Received : July 02, 2024         Accepted : August 20, 2024          Published : September 25, 2024

Abstract

Background: 10-25% of bronchopulmonary sequestration comprises extralobar sequestration (ELS). ELS are usually found between the left lower lobe and diaphragm and rarely infra-diaphragmatically. Case Description: 25 years old young female with 24 weeks of gestation was diagnosed with left supra adrenal mass in fetal anomaly scan. On fetal autopsy, the supra adrenal mass showed lung parenchyma with multiple cystic spaces which are lined by columnar to cuboidal cells. Bronchi and bronchioles were dilated. Skeletal muscle and cartilage were absent. Both grossly and microscopically, lungs and other organs were normal. Diagnosed as extralobar intraabdominal bronchopulmonary sequestration with cystic adenomatoid malformation suggestive of hybrid lesion. Hybrid lesion is a combination of bronchopulmonary sequestration (BPS) and congenital cystic adenomatoid malformation (CCAM). Clinical Relevance: Diagnosis of hybrid lesion during pregnancy is important for prenatal counselling, fetal intervention and birth planning.


References

  • 1.   Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration: report of 7 cases and review of 540 published cases. Thorax1979; 34:96-101.
  • 2.   Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung: a report of 7 cases. J PatholBacteriol1946; 58:457- 67.
  • 3.   D.L. Cass, T.M. Crombleholme, L.J. Howell, P.W. Stafford, E.D. Ruchelli, N. S. Adzick, Cystic lung lesions with systemic arterial blood supply: a hybrid of congenital cystic adenomatoid malformation and bronchopulmonary sequestration, J. Pediatr. Surg1997; 32: 986–90.
  • 4.   Durell J, Thakkar H, Gould S, Fowler D, Lakhoo K. Pathology of asymptomatic, prenatally diagnosed cystic lung malformations. J PediatrSurg2016; 51(2):231–5.
  • 5.   Correia-Pinto J, Gonzaga S, Huang Y, Rottier R.Congenital lung lesions underlying molecular mechanisms. S e m i n P e d i a t r S u r g 2 0 1 0 ; 19(3):171–9.
  • 6.   Yang L, Yang G. Extralobar pulmonary sequestration with a complication of torsion: a case report and literature review. Medicine (Baltimore) 2020; 99(29):e21104.
  • 7.   Ryujin K, Akamine T, Miura N, et al. An adult case of multiple extralobar pulmonary sequestrations in the thoracic and abdominal cavities. Ann ThoracSurg 2022; 113(1):e17-e20.
  • 8.   Petty L, Joseph A, Sanchez J. Case report: Pulmonary sequestration in an adult. Radiol Case Rep 2017; 13(1):21-3.
  • 9.   Raemdonck D, Louw J, et al. Treatment strategies for pulmonary sequestration in childhood: resection, embolization, observation? ActaCardiol 2012; 67:629-34.
  • 10.   David M, Lamas Pinheiro R, Henriques-Coelho T. Prenatal and postnatal management of congenital pulmonary airway malformation. Neonatology 2016; 110:101-15.
  • 11.   Farrugia MK, Raza SA, Gould S, Lakhoo K. Congenital lung lesions: classification and concordance of radiological appearance and surgical pathology. PediatrSurgInt2008; 24: 987–91.
  • 12.   Marshall KW, Blane CE, Teitelbaum DH, van Leeuwen K. Congenital cystic adenomatoid malformation: impact of prenatal diagnosis and changing strategies in the treatment of the asymptomatic patient. AJR Am J Roent- genol2000; 175:1551–4.
  • 13.   Knox EM, Kilby MD, Martin WL, Khan KS. Inutero pulmonary drainage in the management of primary hydrothorax and congenital cystic lung lesion: a systematic review. Ultra sound ObstetGynecol2006; 28:726–34.
  • 14.   Witlox RS, Lopriore E, Oepkes D. Prenatal interventions for fetal lung lesions. PrenatDiagn. 2011; 31(7):628-36.
  • 15.   Loh KC, JelinE, Hirose S, Feldstein V, Goldstein R, Lee H. Microcystic congenital pulmonary airway malformation with hydrops fetalis: steroids vs open fetal resection. J Pediatr Surg 2012; 47:36–39.
  • 16.   Tsao K, Hawgood S, Vu L, Hirose S, Sydorak R, Albanese CT, et al. Resolution of hydrops fetalis in congenital cystic adenomatoid malformation after prenatal steroid therapy. J PediatrSurg2003; 38:508–10.
  • 17.   Curran PF, Jelin EB, Rand L, Hirose S, Feld- stein VA, Goldstein RB, et al.: Prenatal steroids for microcystic congenital cystic adenomatoid malformations. J PediatrSurg 2010; 45:145–50.
  • 18.   Higby K, Melendez BA, Heiman HS: Spontaneous resolution of nonimmune hydrops in a fetus with a cystic adenomatoid malformation. J Perinatol1998; 18:308–10.

Data Sharing Statement

There are no additional data available. All raw data and code are available upon request.

Funding

This research received no funding.

Author Contributions

All authors contributed significantly to the work and approve its publication.

Ethics Declaration

This article does not involve any human or animal subjects, and therefore does not require ethics approval.

Acknowledgements

We would like to express our gratitude to the patients, their families, and all those who have contributed to this study.

Conflicts of Interest

No conflicts of interest in this work.


About this article


Cite this article

Patel S, Pallavi BR, Devi SCS, et al. Hybrid Lesion: Extrapulmonary Sequestration with Cystic Adenomatoid Malformation: A Rare Case Report. Indian J Forensic Med Pathol. 2024;17(3):207-210.


Licence:

Attribution-Non-commercial 4.0 International (CC BY-NC 4.0)

This license enables reusers to distribute, remix, adapt, and build upon the material in any medium or format for noncommercial purposes only, and only so long as attribution is given to the creator.


Received Accepted Published
July 02, 2024 August 20, 2024 September 25, 2024

DOI: https://doi.org/10.21088/ijfmp.0974.3383.17324.9

Keywords

Bronchopulmonary sequestrationHybrid lesionSupra adrenal massFetus

Article Level Metrics

Last Updated

Sunday 21 June 2026, 18:06:21 (IST)


7234

Accesses

10
2068
00

Citations


NA
NA
NA

Download citation


Article Keywords


Keyword Highlighting

Highlight selected keywords in the article text.


Timeline


Received July 02, 2024
Accepted August 20, 2024
Published September 25, 2024

licence


Attribution-Non-commercial 4.0 International (CC BY-NC 4.0)

This license enables reusers to distribute, remix, adapt, and build upon the material in any medium or format for noncommercial purposes only, and only so long as attribution is given to the creator.


Access this article



Share