Abstract
A 78-year-old male who was undergoing treatment for diabetes was referred to our department after a chest X-ray revealed an abnormal shadow in the left upper lung field. A chest contrast-enhanced CT showed a tumor in the left upper lobe and a cystic lesion in the left lower lobe. Two aberrant arteries branching from the aorta were identified near the cyst. The patient was diagnosed with lung cancer and anomalous systemic arterial supply to the basal segment of the left lung (ABLL), necessitating surgical intervention. Thoracoscopic surgery was performed, with the left upper segmentectomy guided by Indocyanine green (ICG) to facilitate precise intersegmental plane identification. The two aberrant arteries were resected, and the perfusion area supplied by these arteries was excised using repeated ICG intravenous injection. This case of lung cancer with ABLL demonstrates that the repeated use of ICG can enable not only a precise upper segmentectomy but also accurate visualization and resection of a perfusion area supplied by aberrant arteries.
Introduction
Anomalous systemic arterial supply to the basal segment of the left lung (ABLL) is a rare congenital vascular anomaly characterized by an aberrant artery originating directly from the aorta. This condition can lead to significant clinical symptoms such as hemoptysis, pneumonia, and right heart failure due to abnormal pulmonary blood flow. Surgical intervention is often required to manage these complications and to resect any affected lung parenchyma. In this report, we present the case of a 78-year-old male diagnosed with lung cancer and ABLL. The patient underwent thoracoscopic surgery that included upper division resection for lung cancer and resection for ABLL of the perfusion area supplied by the anomalous artery originating from the aorta. The use of indocyanine green (ICG) fluorescence imaging, beneficial in thoracic procedures, helped identify intersegmental planes and lung areas perfused by aberrant vessels, enabling precise surgical intervention.
Case
A 78-year-old man was referred to our hospital for the investigation of a lung nodule found during a routine chest radiographic examination. The patient was symptom-free and in good general health. He had diabetes and a history of myocardial infarction. Physical examination did not reveal any remarkable findings, and all laboratory parameters, including HbA1c, were within normal limits. Chest radiography showed an enlarged shadow in the left upper lung field. Chest computed tomography (CT) revealed a 31 mm solid tumor of the left upper lobe and a 15 mm cavity within fluid component by the lung parenchyma of the left lower lobe, close to the Aorta (Figure 1A and 1B). A multiplanar and 3-dimensional reconstruction CT showed a feeding artery branching from the descending aorta near the cavity (Figure 1C). A lung biopsy of the left upper lobe tumor was performed using bronchoscopy, leading to a diagnosis of adenocarcinoma. Positron emission tomography scans showed a significant increase in standard uptake value max to 20 in the left upper lobe tumor, with no evidence of distant metastasis. We made a preoperative diagnosis of a lung cancer (c-T2aN0M0, stageⅠB) and arterial supply to the basal segment of the left lung (ABLL). We performed a left upper segmentectomy and complete excision of the lung area dependent on the anomalous artery containing the cavity, using a thoracoscopic approach with 3 ports. There was adhesion to the surrounding segment 10 and descending aorta. We dissected the adhesion and confirmed two aberrant vessels (Figure 2A). They were carefully dissected and sectioned using an endoscopic stapler (Figure 2B, 2C and 2D). ICG (0.1 mg/kg) was injected into a peripheral vein, and we marked the line between the perfused and non-perfused areas using near-infrared fluorescence imaging (Figure 3A and 3B). Then we performed a resection of the non-perfused area, guided by the marking (Figure 3C). Subsequently, we performed an upper segmentectomy after confirming the absence of metastasis in the hilar lymph node (station 11) during the operation. We performed the intersegmental resection guided by the use of ICG (0.1 mg/kg) after dividing the pulmonary artery, vein and bronchus leading to the upper lobe segment (Figure 3D, 3E and 3F). Histological examination revealed a dilated cystic wall lined with respiratory epithelium, mucous glands, and cartilage. The tumor in the upper division resection was diagnosed as combined small cell carcinoma. The patient recovered uneventfully and was discharged 10 days after the operation. Postoperative chemotherapy was administered and a CT scan two years post-surgery revealed adrenal metastasis, but no pseudoaneurysm was detected near the resected artery in the descending aorta. The patient is currently undergoing treatment for the adrenal metastasis.
Discussion
Arterial supply to the basal segment of the left lung (ABLL) is a relatively rare congenital vascular anomaly. This condition involves a portion of the lung parenchyma, typically supplied by the pulmonary artery, receiving blood flow directly from the aorta, while bronchial anomalies are absent. According to Pryce’s classification, this condition is referred to as type I sequestration [1]. Most patients with ABLL are asymptomatic; however, symptoms such as hemoptysis, recurrent pneumonia, pulmonary hypertension, and heart failure can occur. Given the potential for mortality due to heart failure or massive hemoptysis, treatment is recommended upon diagnosis [2]. Treatment options for ABLL include both surgical and endovascular approaches. Endovascular treatment offers a less invasive alternative to surgery and is particularly suitable for infants [3], but it is not appropriate for cases involving relatively large vascular diameters, and carries the risk of post-treatment pulmonary infarction, necessitating careful patient selection [4]. Surgical methods include the dissection or ligation of the aberrant vessel, with or without lung resection. Several reports indicate that aberrant vessel dissection alone results in symptom improvement and an absence of postoperative complications [5, 6]. Nonetheless, in instances where a large area is perfused by the aberrant vessel, the risk of pulmonary ischemia or infarction may necessitate additional lung resection. Furthermore, supplementary postoperative vascular embolization has been reported for giant aberrant artery [7]. Treatment decisions should therefore be individualized, considering factors such as the perfused region, vessel diameter of aberrant artery, and the presence of symptoms. In the present case, the patient was asymptomatic and elderly, making endovascular treatment a potential option. However, the presence of cystic degeneration posed a risk of future infection or potential malignancy, and the concurrent lesions in the upper lobe warranted a combined surgical approach.
Indocyanine green (ICG) fluorescence imaging technology is employed across various fields. In the thoracic field, it has proven beneficial for identifying intersegmental planes, detecting small pulmonary nodules, assessing blood flow in bronchial stumps and anastomoses, and identifying lymphatic leaks in cases of chylothorax [8]. There are also reports indicating that ICG has been useful in the context of pulmonary resection for ABLL [9, 10]. Compared to existing literature on pulmonary resection for ABLL, our case demonstrates the effective use of ICG to confirm perfusion areas twice, guiding both the pulmonary resection for ABLL and the segmentectomy. Toxicity studies indicate that intravenous injection of ICG at doses up to 5.0 mg/kg is safe [11], although there is a reported 0.05% risk of anaphylactic shock even with ICG doses ranging from 25 to 75 mg [12]. It is advisable, therefore, to use the minimal dose possible to mitigate this risk when administering ICG multiple times. In the present case, despite using a low dose of 0.1 mg/kg, the intersegmental planes were clearly visualized, and ICG was used safely and repeatedly.
The ability to use ICG repeatedly, as demonstrated in this case, is advantageous. Visualizing the perfusion area of the aberrant vessel using ICG could assist in deciding whether to perform vessel dissection alone or in combination with lung resection. Further accumulation of case data is anticipated to enhance this decision-making process. In conclusion, the application of ICG fluorescence imaging offers significant potential for improving the precision of surgical interventions in cases of ABLL. The insights gained from this case underscore the importance of individualized treatment approaches and the utility of advanced imaging techniques in thoracic surgery. Further accumulation of case data will help refine these strategies and optimize patient outcomes.
Conflicts of Interest
None
Availability of Data and Materials
The datasets generated during and/or analyzed during the current study are not publicly available due to contains patient information but are available from the corresponding author on reasonable request.
Ethics Approval and Consent to Participate
We got ethical approval from the ethics committee of Iizuka Hospital, Japan.
Consent for Publication
Written informed consent for the publication of the case details was obtained from our patient.
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