Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2022 Jul 22:10:926942.
doi: 10.3389/fped.2022.926942. eCollection 2022.

Case Report: An extremely rare case of double extralobar pulmonary sequestration with anomalous supplying arteries originating from the abdominal aorta in the left thoracic cavity

Longfei Lv  1   2 , Yunpeng Zhai  1   2 , Huashan Zhao  1   2 , Rui Guo  1   2 , Hongxiu Xu  1   2 , Shisong Zhang  1   2
Affiliations
Case Reports

Case Report: An extremely rare case of double extralobar pulmonary sequestration with anomalous supplying arteries originating from the abdominal aorta in the left thoracic cavity

Longfei Lv et al. Front Pediatr. .

Abstract

To the best of our knowledge, double or multiple extralobar pulmonary sequestrations (PSs) with anomalous arterial supply in the ipsilateral thoracic cavity have rarely been reported before. PS can be divided into two types: intralobar sequestration (ILS) and extralobar sequestration (ELS). We encountered a 5-month-old infant with double ELS in the left thoracic cavity that was incidentally detected during thoracoscopic surgery. Surgical exploration revealed two separate, well-circumscribed abnormal masses in the left thoracic cavity, and the patient was successfully treated using thoracoscopic surgery. Postoperative pathology confirmed that both masses were PS tissues. Accurate preoperative diagnosis using CT alone may be inadequate in this type of case. Therefore, thoracoscopy may be more suitable for diagnosing and treating unusual ELS.

Keywords: congenital lung malformation; extralobar sequestration; intralobar sequestration; pulmonary sequestration; thoracoscopic surgery.

PubMed Disclaimer

Conflict of interest statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Figures

FIGURE 1
FIGURE 1
The enhanced chest CT of the case before surgery. (A) CT images in the AP showed two areas with different enhancement degrees in one mass, and the upper pole of the mass was adjacent to the inferior lobe of the left lung. (B) CT images in the AP showed that the largest cross-section of the mass was close to the upper pole of the spleen. (C) CT images in the AP showed the origin of the upper group of anomalous vessels (referring back to this image postoperatively, we found that it showed the origin of the anomalous supplying arteries and a draining vein of the upper ELS). (D) CT images in the AP showed the origin of the inferior group of anomalous vessels (referring back to this image postoperatively, we found that it showed the origin of an anomalous supplying artery of the inferior ELS). The draining vein of the inferior lesion cannot be distinguished. CT, computed tomography; AP, arterial phase; ELS, extralobar sequestration. ◼: upper ELS; ▲: inferior ELS; ★: spleen; ◆: draining vein of the upper ELS; ▼: the origin of anomalous supplying arteries of the upper ELS; ⚫: anomalous vessels in the left diaphragm connecting to the inferior ELS; ↑: the origin of an anomalous supplying artery of the inferior ELS.
FIGURE 2
FIGURE 2
Intraoperative findings. (A) The inferior ELS was adjacent to the left diaphragm, and the base of the inferior ELS was closely related to the left diaphragm. (B) The inferior ELS was resected after double ligation of the anomalous vessels using two absorbable vessel clamps. (C) The upper ELS was adjacent to the left lower pulmonary ligament. Four anomalous vessels connecting to the upper ELS could be seen. (D) The upper ELS was resected after double ligation of the anomalous vessels using two absorbable vessel clamps. ELS, extralobar sequestration.
FIGURE 3
FIGURE 3
Resected pathological tissue. The pink tissue on the left of the image is the inferior ELS, which was approximately 3.7 cm × 2.7 cm × 2 cm, and the dark red tissue on the right of the image is the upper ELS, which was approximately 3.8 cm × 2.9 cm × 2.5 cm. ELS, extralobar sequestration.
FIGURE 4
FIGURE 4
Histopathological findings. (A) Histopathological imaging of the inferior ELS. The tissue is mainly composed of abnormal airway epithelium, which contains fewer arterial and venous components with smaller diameters (hematoxylin and eosin stain, × 10). (B) Histopathological imaging of the upper ELS. The tissue is characterized by numerous arterial and venous components with larger diameters (hematoxylin and eosin stain, × 10).
See this image and copyright information in PMC

References

    1. Ryujin K, Akamine T, Miura N, Yoshida R, Shikada Y, Kato S, et al. An adult case of multiple extralobar pulmonary sequestrations in the thoracic and abdominal cavities. Ann Thorac Surg. (2022) 113:e17–20. 10.1016/j.athoracsur.2021.03.083 - DOI - PubMed
    1. Taylor JA, Laor T, Warner BW. Extralobar pulmonary sequestration. Surgery. (2008) 143:833–4. 10.1016/j.surg.2007.06.012 - DOI - PubMed
    1. Jeanfaivre T, Afi M, L’hoste P, Tuchais E. Simultaneous discovery of bilateral intralobar and extralobar pulmonary sequestrations. Ann Thorac Surg. (1997) 63:1171–3. 10.1016/s0003-4975(97)00070-2 - DOI - PubMed
    1. Woerner A, Schwendener K, Wolf R, Nelle M. Neonatological and pulmonological management of bilateral pulmonary sequestration in a neonate. World J Pediatr. (2008) 4:301–4. 10.1007/s12519-008-0054-2 - DOI - PubMed
    1. Urushihara N, Nakagawa Y, Kawashima K, Fukazawa T, Watanabe Y, Todani T. Congenital right diaphragmatic hernias through posterolateral and anterolateral defects associated with extralobar pulmonary sequestration: a case report. J Pediatr Surg. (1997) 32:101–2. 10.1016/s0022-3468(97)90106-8 - DOI - PubMed

Publication types

LinkOut - more resources