Case Report Open Access
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 26, 2021; 9(27): 8192-8198
Published online Sep 26, 2021. doi: 10.12998/wjcc.v9.i27.8192
Surgical treatment of abnormal systemic artery to the left lower lobe: A case report
Yi-Yuan Zhang, Xiao-Ying Gu, Jia-Lin Li, Zhao Liu, Guo-Yue Lv
Yi-Yuan Zhang, Jia-Lin Li, Zhao Liu, Department of Thoracic Surgery, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China
Xiao-Ying Gu, Department of Anesthesiology, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China
Guo-Yue Lv, Department of Hepatobiliary and Pancreatic Surgery, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China
ORCID number: Yi-Yuan Zhang (0000-0002-9088-7228); Xiao-Ying Gu (0000-0002-7124-7428); Jia-Lin Li (0000-0002-8541-2797); Zhao Liu (0000-0003-0451-4693); Guo-Yue Lv (0000-0001-9115-5945).
Author contributions: Zhang YY performed the study and wrote the paper; Gu XY provided technical advice and co-wrote the article; Li JL and Liu Z collected clinical data; Lv GY designed and supervised the study; All authors have read and approved the manuscript.
Informed consent statement: Written informed consent was obtained from the patient for publication of this case report.
Conflict-of-interest statement: The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Guo-Yue Lv, PhD, Chief Doctor, Department of Hepatobiliary and Pancreatic Surgery, The First Hospital of Jilin University, No. 71 Xinmin Street, Changchun 130021, Jilin Province, China. lvgy@jlu.edu.cn
Received: April 23, 2021
Peer-review started: April 23, 2021
First decision: June 6, 2021
Revised: June 30, 2021
Accepted: August 3, 2021
Article in press: August 3, 2021
Published online: September 26, 2021

Abstract
BACKGROUND

Abnormal systemic artery to the left lower lobe is a rare congenital abnormality characterized by anomalous communication between the systemic and pulmonary circulation. Owing to its rarity, there is limited clinical experience with respect to the diagnosis and treatment of this disease.

CASE SUMMARY

We report a 60-year-old man who presented with a history of hemoptysis for 20 d. Contrast-enhanced computed tomography of the chest confirmed the diagnosis of abnormal systemic artery to the left lower lobe, and surgical treatment was performed. The aberrant artery arising from the descending thoracic aorta was ligated, followed by removal of the left lower lobe. The patient showed good recovery and was discharged 6 d after the surgery. At the 1-year follow-up, the patient had recovered completely, and lung CT showed no abnormal findings.

CONCLUSION

We present a case of abnormal systemic artery to the left lower lobe that was successfully managed by surgical resection of the aberrant artery and the left lower lobe. This case report adds to the clinical experience of diagnosing and treating this rare entity.

Key Words: Abnormal systemic artery to the left lower lobe, Diagnosis and treatment, Surgery, Rare disease, Computed tomography, Case report

Core Tip: Abnormal systemic artery to the left lower lobe is a rare congenital vascular malformation. Experience in diagnosis and treatment remains limited. Here we presented the clinical presentation, contrast-enhanced computed tomography, surgical procedure and follow-up result of an abnormal systemic artery to the left lower lobe case. We discussed the difference between abnormal systemic artery to the left lower lobe and pulmonary sequestration and different treatment strategies. This case report adds to the clinical experience with regard to the diagnosis and treatment of this rare disease.



INTRODUCTION

Abnormal systemic artery to the left lower lobe (ASALLL) is a rare congenital anomaly that creates an anomalous communication between the systemic and the pulmonary circulation[1]. It is characterized by aberrant systemic arterial blood flow to the left lower lobe of the lung, which drains into the pulmonary vein[1,2]. Owing to its rarity, only a few reports have described this entity. Hemoptysis is the most common manifestation, while some patients may exhibit no symptoms; in rare cases, patients may develop fever and cough with or without expectoration[2-4]. Surgical resection or therapeutic embolization are the main treatment modalities, while some cases may require vascular reconstruction[2,5,6]. We report a senile patient with ASALLL who was successfully treated by surgical resection. In addition, we summarize the diagnosis and treatment of ASALLL based on literature review.

CASE PRESENTATION
Chief complaints

A 60-year-old man was admitted to our hospital in October 2019 with a chief complaint of hemoptysis in the past 20 d. There was no obvious cause of hemoptysis, and it was not accompanied by any other symptoms like chest pain, cough, expectoration or fever.

History of present illness

The symptoms started 20 d ago with no obvious cause of hemoptysis.

History of past illness

The past history was unremarkable apart from a history of appendicectomy. The patient was otherwise healthy and had no history of hypertension, diabetes mellitus, coronary heart disease or infectious disease.

Personal and family history

The patient is a life-long non-smoker. He denied any family history of related diseases.

Physical examination

The general condition of the patient at presentation was good. His vital signs were normal with stable hemodynamic status. Breath sounds were clear, and no pulmonary crackles were detected on auscultation.

Laboratory examinations

The laboratory parameters of the patient were almost normal.

Imaging examinations

Pulmonary contrast-enhanced computed tomography (CT) with three-dimensional reconstruction performed immediately after admission revealed an aberrant vessel supplying the left lower lung lobe (Figure 1); its origin was traced to the descending thoracic aorta. At the same time, pulmonary artery of the left lower lobe of the lung was absent. There were no abnormalities in the left lower pulmonary vein or in the bronchus of the left lower lobe of the lung.

Figure 1
Figure 1 Contrast-enhanced computed tomography scan images with three-dimensional imaging. The yellow arrows show the abnormal systemic artery. The green arrow shows the normal left inferior lobar bronchus. The blue arrow shows the left superior lobe pulmonary artery and its branches, and the left lower lobe pulmonary artery is missing.
FINAL DIAGNOSIS

The potential differential diagnoses in this case were ASALLL and pulmonary sequestration (PS). The corresponding pulmonary artery is usually absent in ASALLL, but the bronchus is usually normally developed. PS is characterized by the absence of normal communication with the bronchial tree, which helps differentiate it from ASALLL. The final diagnosis of the presented case was ASALLL.

TREATMENT

Routine preoperative workup did not reveal any other abnormality, and the patient underwent surgical treatment in November 2019. After general anesthesia and intubation with double lumen tube, the thoracic cavity was opened via the fifth intercostal space. An anomalous systemic artery arising from the thoracic aorta and supplying blood to the left lower lobe was identified during surgery; the anomalous artery was ligated by vascular stapler and subsequently sutured. The pulmonary artery supplying the left lower lobe was absent, while the corresponding pulmonary artery and bronchus were normal and were all sundered during the removal of the left lower lobe. The surgery was uneventful, and the thoracic cavity was closed after the placement of a closed drainage tube.

OUTCOME AND FOLLOW-UP

The patient showed good recovery and was discharged after removal of the drainage tube 6 d after the surgery. At the 1-year follow-up, the patient had recovered completely, and no abnormalities were detected on lung CT.

DISCUSSION

ASALLL is a rare congenital arterial malformation that is relatively more common in the Asian population[7]. Pathologically, an aberrant artery originates from the descending aorta and supplies blood to the left lower lobe. It is usually associated with the absence of the corresponding pulmonary artery, but the bronchus is normal. It is believed that insufficient blood supply to the lung tissue during embryo development stimulates the abnormal branching of the systemic artery. ASALLL needs to be differentiated from another rare congenital disease, PS, which is characterized by nonfunctional pulmonary tissue that lacks normal communication with the bronchial tree and is also supplied by a non-pulmonary systemic artery[8]. ASALLL was previously categorized as Pryce type Ⅰ pulmonary sequestration; however, this categorization was contested due to the presence of normal bronchus in the affected pulmonary tissue[4]. ASALLL also differs from PS in terms of clinical manifestations, as respiratory symptoms are more common in patients with ASALLL due to the normal communication between the involved lung tissue and the airway.

We conducted a literature search in the PubMed database for all papers written in English utilizing the search term: “Abnormal Systemic Artery to the Left Lower Lobe.” A total of 82 previous cases published since 1976 were retrieved. A summary of the 83 cases (including the present case) is presented in Table 1. Among these, 59% of patients (48 cases) manifested with hemoptysis, 8% (7 cases) with tachypnea or dyspnea, 6% (5 cases) with cough, 4% patients (3 cases) with chest pain and 33% patients (27 cases) were asymptomatic. Hemoptysis was the most commonly reported manifestation of ASALLL and was also the sole symptom in the present case.

Table 1 Summary of previously reported cases of abnormal systemic artery to the left lower lobe published since 1976.
Yr
Author
n
Age in yr
Symptoms
Treatment
Follow-up
Complication
1976Kirks 26 moAsymptomaticLobectomy--
17 moAsymptomaticLobectomy
1979Ishihara15AsymptomaticLobectomy--
1981Yabek16 moTachypneaLobectomy6 moNone
1992Tao138DyspneaLobectomy--
1993Kurosaki359Hemoptysis---
54AsymptomaticLobectomy
46AsymptomaticSegmentectomy
1996Hirai229HemoptysisLobectomy6 moNone
30AsymptomaticAnastomosis
1999Yamanka246CoughLobectomy--
68Dyspnea, hemoptysisSegmentectomy
2000Ko339HemoptysisLobectomy9 moNone
58AsymptomaticNone6 moNone
47Pain, hemoptysisNone2 yrChest pain
2001Ashizawa529AsymptomaticSurgery (5)--
Asymptomatic
Asymptomatic
Dyspnea
Hemoptysis
2002Kim1149Asymptomatic (11)None (11)--
2003Iizasa450AsymptomaticSegmentectomy --
17HemoptysisSegmentectomy
43HemoptysisSegmentectomy
20AsymptomaticLobectomy
2004Haraguchi129HemoptysisLobectomy--
2006Saida141HemoptysisEmbolization6 yrChest pain for 1 mo
2008Albertini121Dyspnea, hemoptysisLobectomy--
2008Wu124HemoptysisNone2.5 yrHemoptysis
2009Wong110 moTachypneaLobectomy--
2009Lim126Hemoptysis, chest painEmbolization6 moNone
2011Jiang433HemoptysisEmbolization2 wk2 patients with chest pain for 2 wk
28HemoptysisEmbolization
24HemoptysisEmbolization
19HemoptysisEmbolization
2011Abe124HemoptysisEmbolization1 yrNone
2011Canyigit153Chest painEmbolization10 moChest pain for 1 wk
2012Anil123HemoptysisEmbolization6 moChest discomfort for 2 d
2013Higuchi158AsymptomaticSegmentectomy--
2013Sugihara167FatigueEmbolization9 moNone
2015Kawaguchi125HemoptysisLigation4 yrNone
2015Qin1623Hemoptysis (16), Cough (4), Sputum (2)---
2016Jiang9132.7Hemoptysis (9)Embolization (9)42.1 mo4 patients with chest pain for weeks
2017Miller12 moTachypneaAnastomosis6 wkNone
2018Thakur118HemoptysisLigation6 wkNone
2018Kim17 moAsymptomaticAnastomosis3 yrNone
2020Utsumi142AsymptomaticLobectomy6 moNone
2020Hu153HemoptysisMedical treatment--
2021Ema139AsymptomaticSegmentectomy2 moNone
2021Current case160HemoptysisLobectomy1 yrNone

The blood pressure in the systematic circulation is considerably higher than that in the pulmonary circulation; therefore, perfusion of pulmonary tissue by the systematic circulation can result in alveolar bleeding, leading to hemoptysis[9]. As hemoptysis can also occur in PS[10], the differentiation between ASALLL and PS relies on further investigations. Contrast-enhanced chest CT with three-dimensional reconstruction is the main modality for diagnosis of ASALLL, as it can easily delineate the aberrant artery supplying the left lower lobe[1]. However, differentiation of ASALL from PS based on CT findings can be challenging, as both conditions exhibit similar arterial malformation. In a study, the diameter of the abnormal artery of ASALLL was significantly greater than that of PS (11.88 ± 1.13 mm vs 5.96 ± 0.98 mm), which can facilitate differentiation between the two conditions on CT[11]. Moreover, patients with ASALLL commonly exhibit ground-glass opacity on CT, which is attributable to alveolar bleeding; in contrast, the typical manifestation of PS on CT scan is solid or solid-cystic mass, which is attributable to coinfection[11].

Persistent communication between the systematic circulation and the pulmonary circulation in the pulmonary tissue results in a series of consequences such as alveolar hemorrhage, increase in cardiac load and impaired oxygenation. The therapeutic goal for this disease is to correct the left-to-right shunt, which can be achieved by pulmonary lobar or segmental resection, embolism or ligation of the aberrant artery and anastomosis between the abnormal systemic artery and left pulmonary artery[2,12,13]. All these therapeutic techniques have been applied in the clinical practice. Our retrospective analysis of previous cases showed that 39% of patients underwent surgical treatment (15 cases lobectomy, 7 cases segmentectomy, 3 cases anastomosis, 2 cases ligation), 23% of patients (19 cases) were treated with embolization, and 17% of patients (14 cases) did not receive any treatment. No complications were reported in patients who underwent surgery, while 47% of patients (9 cases) who underwent embolization developed symptoms of chest pain lasting for weeks (Table 1). Some patients developed chest pain after artery embolism, which was attributable to pulmonary infarction, as identified on postoperative CT scan[14]. Inadequate tissue perfusion was also reported in patients who underwent anastomosis, which is likely attributable to the vascular damage in the aberrant artery caused by prolonged high-pressure blood flow[15,16]. Thus, anastomosis is preferred in asymptomatic pediatric patients, as these patients tend to have better vascular status and can easily endure this procedure. However, pulmonary resection or artery embolism are recommended for senile patients, especially those who present with hemoptysis[2]. Owing to the limited clinical experience with this disease, further studies are required to identify optimal treatment for this rare disease.

CONCLUSION

ASALLL is a rare congenital vascular malformation with limited knowledge about its diagnosis and treatment. Our experience in the management of ASALLL highlights the importance of contrast-enhanced computed tomography in the diagnosis of this disease. Specifically, ASALLL needs to be differentiated from PS as these two conditions require different treatment strategies. This case report adds to the clinical experience of this rare disease.

Footnotes

Manuscript source: Unsolicited manuscript

Specialty type: Surgery

Country/Territory of origin: China

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P-Reviewer: Naseer M S-Editor: Zhang H L-Editor: Filipodia P-Editor: Yuan YY

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