Sapna Patel Associate Professor, Department of Pathology, JSS Academy of Higher Education and Research, Mysuru 570 015, Karnataka, India
Pallavi BR Senior Resident, Department of Pathology, JSS Academy of Higher Education and Research, Mysuru 570 015, Karnataka, India
Sheela Devi CS Professor and Head of Department, Department of Pathology, JSS Academy of Higher Education and Research, Mysuru 570 015, Karnataka, India
Devi Sreelakshmi Post graduate, Department of Pathology, JSS Academy of Higher Education and Research, Mysuru 570 015, Karnataka, India
Address for correspondence: Pallavi BR, Senior Resident, Department of Pathology, JSS Academy of Higher Education and Research, Mysuru 570 015, Karnataka, India E-mail: pallubr@gmail.com
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.
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.
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.
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.
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.
Description: Fig. 1-A –Left supra adrenal mass in fetal anomaly scan with vascular supply from aorta
Heading
Description: Fig. 2: Microscopy showed lung parenchyma with multiple cystic spaces lined by cuboidal to columnar cells. Bronchi and bronchioles were dilated