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CASE REPORT Open Access Intralobar pulmonary sequestration displayed as localized emphysema on computed tomography image Weibo Qi * , Junjie Zhao, Guping Shi and Fan Yang Abstract Background: Pulmonary sequestration is a relatively rare condition in which a systemic artery supplies blood to an abnormal lung tissue and the normal connection with the bronchial tree is absent. It can be displayed as various signs on the computed tomography image, but emphysema is extremely rare. Case presentation: We describe the case of a 35-year-old man with intralobar pulmonary sequestration that appeared as localized emphysema on the computed tomography image. The 3-D reconstruction revealed the presence of an anomalous feeding artery and the absence of normal connection with the bronchial tree. Conclusion: We presumed that it was a type between anomalous systemic arterial supply to the normal lungand the common type of pulmonary sequestration. Common pulmonary lobectomy was performed and the patient recovered well. Keywords: Pulmonary, Sequestration, Computed tomography Background In pulmonary sequestration, the lung lesion is usually serious, and the pulmonary alveoli have completely lost their normal structure due to the lack of a normal con- nection with the bronchial tree. Herein, we report a case of pulmonary sequestration in which the lung lesion was relatively slight and was shown as localized emphysema on the computed tomography image. Case presentation The patient, a 35-year-old man who coughed for 1 week was referred to our hospital. He had no history of smoking, and the physical examination and the laboratory investiga- tion showed no particular findings. The computed tomog- raphy scan of the chest revealed emphysema localized in the posterior basal segment of the right lower pulmonary lobe. The further three-dimensional computed tomography image showed that the anomalous feeding artery originated from the celiac trunk. These findings led us to the diagnosis of pulmonary sequestration and because the normal function of respir- ation was absent in the lesion, we decided to remove the right lower lobe. The thoracoscopic lobectomy of the lung confirmed the existence of an anomalous feeding artery. The patient recovered well and was discharged from the hospital on postoperative day 7. No problems were observed during the 4-year postoperative follow-up period. Discussion Pulmonary sequestration accounts for 0.156.40% of all congenital lung malformations [1]. Usually, the condition is difficult to diagnose preoperatively because of the vari- ous clinical manifestations presented. Pryce [2] described this disease in 1946. Pulmonary sequestration is divided into two types: intralobar and extralobar. It is noteworthy that Pryce [2] subdivided intralobar pulmonary seques- tration into three types. According to his classification, in type 1, there is no abnormal lung tissue. Since no lung sequestration is present in this type, many profes- sionals have recently named it anomalous systemic arterial supply to the normal lungto distinguish it from the real pulmonary sequestration. Therefore, * Correspondence: [email protected] Department of Cardio-Thoracic Surgery, The First Hospital of Jiaxing, Jiaxing, 314000 Zhejiang, Peoples Republic of China © The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Qi et al. Journal of Cardiothoracic Surgery (2017) 12:83 DOI 10.1186/s13019-017-0646-9

Intralobar pulmonary sequestration displayed as localized ......Lower accessory pulmonary artery with intralobar sequestration of lung; a report of seven cases. J Pathol Bacteriol

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Page 1: Intralobar pulmonary sequestration displayed as localized ......Lower accessory pulmonary artery with intralobar sequestration of lung; a report of seven cases. J Pathol Bacteriol

CASE REPORT Open Access

Intralobar pulmonary sequestrationdisplayed as localized emphysema oncomputed tomography imageWeibo Qi*, Junjie Zhao, Guping Shi and Fan Yang

Abstract

Background: Pulmonary sequestration is a relatively rare condition in which a systemic artery supplies blood to anabnormal lung tissue and the normal connection with the bronchial tree is absent. It can be displayed as varioussigns on the computed tomography image, but emphysema is extremely rare.

Case presentation: We describe the case of a 35-year-old man with intralobar pulmonary sequestration thatappeared as localized emphysema on the computed tomography image. The 3-D reconstruction revealed thepresence of an anomalous feeding artery and the absence of normal connection with the bronchial tree.

Conclusion: We presumed that it was a type between “anomalous systemic arterial supply to the normallung” and the common type of pulmonary sequestration. Common pulmonary lobectomy was performed andthe patient recovered well.

Keywords: Pulmonary, Sequestration, Computed tomography

BackgroundIn pulmonary sequestration, the lung lesion is usuallyserious, and the pulmonary alveoli have completely losttheir normal structure due to the lack of a normal con-nection with the bronchial tree. Herein, we report a caseof pulmonary sequestration in which the lung lesion wasrelatively slight and was shown as localized emphysemaon the computed tomography image.

Case presentationThe patient, a 35-year-old man who coughed for 1 weekwas referred to our hospital. He had no history of smoking,and the physical examination and the laboratory investiga-tion showed no particular findings. The computed tomog-raphy scan of the chest revealed emphysema localized inthe posterior basal segment of the right lower pulmonarylobe. The further three-dimensional computed tomographyimage showed that the anomalous feeding artery originatedfrom the celiac trunk.

These findings led us to the diagnosis of pulmonarysequestration and because the normal function of respir-ation was absent in the lesion, we decided to remove theright lower lobe. The thoracoscopic lobectomy of thelung confirmed the existence of an anomalous feedingartery. The patient recovered well and was dischargedfrom the hospital on postoperative day 7. No problemswere observed during the 4-year postoperative follow-upperiod.

DiscussionPulmonary sequestration accounts for 0.15–6.40% of allcongenital lung malformations [1]. Usually, the conditionis difficult to diagnose preoperatively because of the vari-ous clinical manifestations presented. Pryce [2] describedthis disease in 1946. Pulmonary sequestration is dividedinto two types: intralobar and extralobar. It is noteworthythat Pryce [2] subdivided intralobar pulmonary seques-tration into three types. According to his classification,in type 1, there is no abnormal lung tissue. Since nolung sequestration is present in this type, many profes-sionals have recently named it “anomalous systemicarterial supply to the normal lung” to distinguish itfrom the real pulmonary sequestration. Therefore,

* Correspondence: [email protected] of Cardio-Thoracic Surgery, The First Hospital of Jiaxing, Jiaxing,314000 Zhejiang, People’s Republic of China

© The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Qi et al. Journal of Cardiothoracic Surgery (2017) 12:83 DOI 10.1186/s13019-017-0646-9

Page 2: Intralobar pulmonary sequestration displayed as localized ......Lower accessory pulmonary artery with intralobar sequestration of lung; a report of seven cases. J Pathol Bacteriol

pulmonary sequestration is a condition in which asegment or lobe of lung tissue has no bronchial com-munication with the normal tracheobronchial tree.Localized emphysema occurs rarely (Fig. 1), and on a

computed tomographic image, the abnormal lung tissuecould be displayed as any of the following: a cystic lesion,mass, lamellar shadow, bronchiectasis, encapsulatedhydrothorax, or atelectasis. In the case described in thepresent study, the lung lesion was slighter than that in the

common type of pulmonary sequestration. Thus, we pre-sumed that it was a type between the previously defined“anomalous systemic arterial supply to the normal lung”and the common type of pulmonary sequestration. Im-portantly, the finding of the anomalous feeding artery ledus to the right diagnosis. It was clearly visible on the com-puted tomography image that the feeding artery enteredthe abnormal lung tissue (Fig. 2). The three-dimensionalcomputed tomographic image showed the full view of(Fig. 3). The sagittal view image revealed the absence of

Fig. 1 Emphysema localized on the posterior basal segment of theright lobe and the anomalous feeding artery (arrow) located in theabnormal lung tissue

Fig. 2 The feeding artery (arrow) enters the right lower lobe ofthe lung

Fig. 3 A three-dimensional reconstruction image showing the fullview of the feeding artery (arrow) originating from the celiac trunk

Fig. 4 No normal branch of the tracheobronchial tree to the rightposterior basal segment was present

Qi et al. Journal of Cardiothoracic Surgery (2017) 12:83 Page 2 of 3

Page 3: Intralobar pulmonary sequestration displayed as localized ......Lower accessory pulmonary artery with intralobar sequestration of lung; a report of seven cases. J Pathol Bacteriol

the normal branch of the tracheobronchial tree to the pos-terior basal segment of the right lobe (Fig. 4).Conventionally, angiography has been the gold standard

for the diagnosis of pulmonary sequestration. However,multiplanar CT and 3-dimensional reconstruction havebeen increasingly replacing this diagnostic method due totheir lower invasiveness, better spatial resolution indepicting the vessel anatomy of the sequestrated lungtissue [3, 4], and reduced risk of unexpected bleeding inoperation. Although the employment of coil embolizationas a promising technique has been reported, some au-thors, such as Kohei Ando [5], consider it just a solutionto deal with the patient of “anomalous systemic arterialsupply to the normal lung”.

ConclusionAt present, pulmonary lobectomy is the most commonsurgical treatment for pulmonary sequestration. The em-ployment of coil embolization is just a solution to thepatient of “anomalous systemic arterial supply to thenormal lung”. Furthermore, we also deem that the exci-sion of the abnormal lung lobe is reasonable because ithas lost the function of respiration.

AcknowledgementsNone.

FundingThis study was supported by the Jiaxing Thoracic Surgery Key DisciplineFund (04-F-17).

Availability of data and materialsAll data analyzed during this study are included in this published article.

Authors’ contributionsWBQ, JJZ and GPS were involved in patient care. WBQ and FY were involvedin manuscript preparation and revisions. All authors read and approved thefinal manuscript.

Ethics approval and consent to participateEthics Committee of The First Hospital of Jiaxing approved this case report. Acopy of approval letter is available for review by the Editor of this journal.

Consent for publicationWritten informed consent was obtained from the patient’s wife forpublication of this case report and any accompanying images. A copy of thewritten consent is available for review by the Editor of this journal.

Competing interestsThe authors declare that they have no competing interests.

Publisher’s NoteSpringer Nature remains neutral with regard to jurisdictional claims inpublished maps and institutional affiliations.

Received: 29 March 2017 Accepted: 29 August 2017

References1. Kestenholz PB, Schneiter D, Hillinger S, Lardinois D, Weder W.

Thoracoscopic treatment of pulmonary sequestration. Eur J CardiothoracSurg. 2006;29(5):815–8.

2. Pryce DM. Lower accessory pulmonary artery with intralobar sequestrationof lung; a report of seven cases. J Pathol Bacteriol. 1946;58(3):457–67.

3. Platon A, Poletti PA. Image in clinical medicine. Pulmonary sequestration. NEngl J Med. 2005;353(20):e18.

4. Abbey P, Das CJ, Pangtey GS, Seith A, Dutta R, Kumar A. Imaging inbronchopulmonary sequestration. J Med Imaging Radiat Oncol. 2009;53(1):22–31.

5. Ando K, Maehara T, Adachi H, Konishi T, Fukata M, Furukawa H, et al.Intralobar pulmonary sequestration supplied by an anomalous aneurysmalartery. Ann Thorac Surg. 2012;93(1):319–22.

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Qi et al. Journal of Cardiothoracic Surgery (2017) 12:83 Page 3 of 3