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Single-port thoracoscopic removal of an azygos vein aneurysm: a case report and literature review

Single-port thoracoscopic removal of an azygos vein aneurysm: a case report and literature review Background Azygos vein aneurysms (AVAs) are extremely rare. The majority of patients have no obvious clinical symptoms, so they are found by physical examination or by chance. There is limited clinical treatment experience that can be referred to, and there are no clear guidelines or research evidence standardizing the surgical and interven- tional therapy. Here, we report a patient with idiopathic AVA whose three-dimensional reconstruction of the tumor was completed before surgery. On the basis of three-dimensional reconstruction, single-port thoracoscopic resection of the AVA was successfully completed and reported for the first time. The previously reported cases are summarized to provide guidance for the diagnosis and treatment of patients with AVAs. Case presentation A 56-year-old man was transferred to our hospital due to “dysphagia”. The diagnosis of AVA was made after enhanced computed tomography, gastroscopy, fiberoptic bronchoscopy, and three-dimensional recon- struction. Congenital weakness or degenerative changes causes the vein walls to be extremely thin that the AVA had the risk of ruptur. Furthermore, the patient had symptoms of dysphagia, he received single-port thoracoscopic surgery. After the operation, his dysphagia disappeared. The postoperative pathology confirmed hemangioma. The patient was discharged 3 days after surgery without any complications. Conclusions AVAs are rare. Preoperative three-dimensional reconstruction can greatly help surgeons clarify the disease diagnosis, formulate the surgical plan, avoid damage to the surrounding vital organs, and reduce intraopera- tive bleeding. Thoracoscopic surgery to remove AVAs is difficult and has a high risk of bleeding, while more minimally invasive single-port thoracoscopic surgery is also safe and effective for the treatment of AVAs. Keywords Azygos vein aneurysm, Hemangioma of the azygos vein arch, Single-port thoracoscopic surgery, Three- dimensional reconstruction, Case report Background Yi Yao and Qiuxia Hu contributed to this work equally and thus are co-first authors Azygos vein aneurysms (AVAs) are rare [1]. In radiologic *Correspondence: studies, the normal diameter of the AV is defifined as no Ji Li more than 1  cm [2]. Underlying causes of AVA forma- njyyxxwklcy@163.com 1 tion that have been proposed include cardiac decompen- Department of Cardiothoracic Surgery, The First People’s Hospital of Neijiang, No. 1866, West Section of Hanan Avenue, Shizhong District, sation [3, 4], portal hypertension [4, 5], pregnancy [3], Neijiang 641000, Sichuan, China and compression of the SVC due to neoplasms [3, 5]. It Department of Obstetrics and Gynecology, The First People’s Hospital may occur rupture, thromboembolism, mediastinal mass of Neijiang, No. 1866, West Section of Hanan Avenue, Shizhong District, Neijiang 641000, Sichuan, China effects, and pulmonary artery hypertension with progres - sive gradual enlargement of the AVA [3]. Because most © The Author(s) 2023. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. The Creative Commons Public Domain Dedication waiver (http:// creat iveco mmons. org/ publi cdoma in/ zero/1. 0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 2 of 9 patients are asymptomatic, the diagnosis is mostly an enhancement in the arterial phase. It showed delayed incidental finding. The optimal management of AVAs enhancement with a CT value of approximately 118 HU remains uncertain, and there is no clear distinction of in the venous phase (Fig.  1A). Bronchoscopy revealed criteria indicating conservative and surgical or interven- external pressure stenosis at the bronchial origin of the tional therapy. Usually, surgical resection is required in left lower lobe (Fig.  1B). Gastroscopy showed no obvi- cases of compression of adjacent structures, appearance ous abnormalities. The initial diagnosis was hemangioma of clinical symptoms, azygos vein (AV) thrombus for- or bronchial cyst. Three-dimensional reconstruction mation in patients with oral anticoagulation, contrain- showed that the mass was connected to the AV and supe- dication to oral anticoagulation, pulmonary embolism, rior vena cava. The mass body obviously extruded into and considerable increase in the diameter of an AVA the esophagus and trachea. There were two arteries from [1]. Due to the high difficulty of thoracoscopic surgi - the aorta running among the mass body, the esophagus cal resection and the risk of embolism migration. Most and the trachea (Fig.  1C, D). These findings revealed patients underwent thoracotomy to remove AVAs, and that the mass is considered to be initial diagnosed as a only a few cases reported successful thoracoscopic resec- hemangioma originating from the azygos vein arch. Sur- tion [1]. There is no report of single-port thoracoscopic gical resection should take care with the aortic branch resection of an AVA. On the basis of three-dimensional behind the AVA to prevent uncontrollable bleed. At the reconstruction, we report a case of single-port thora- same time, we need to protect the vagus nerve and the coscopic-based management of an idiopathic AVA with thoracic duct. Avoid damaging them to prevent gastroin- symptoms of dysphagia. At the same time, we review the testinal symptoms and chylothorax. symptoms, diagnosis, and treatment of AVAs in the pub- Dysphagia caused by AVA extruded the esophagus and lished literature. the left bronchus. Interventional treatment may can’t cure dysphagia symptom. In addition, interventional Case report treatment may not prevent future migration and embo- A 56-year-old man complained of dysphagia. He was lization of the thrombus. So, we chose single-port video- referred to our hospital without special treatment assisted thoracoscopic surgery (VATS) to respect the because of a mediastinal mass on chest computed tomog- AVA under double-lumen tracheal intubation 3 days after raphy (CT). The patient had no relevant medical history. hospitalization. The patient was in the left lateral decu - He denied any falls or other trauma. Body temperature bitus position during surgery. The intraoperative explora - was 37.0 °C, heart rate was 78 beats/min, blood pressure tion results were consistent with the three-dimensional was 127/74 mmHg, and respiratory rate was 20 breaths/ reconstruction. The AVA was located in the azygos vein min. Breath sounds were normal. No moist or dry rales. arch, and there was no abnormal pulmonary artery or No heart murmurs or muffled heart tones. Contrast- pulmonary vein connected with it (Fig.  2A). The AVA enhanced CT revealed a mediastinal mass (4.2 × 3.7 obviously extruded into the esophagus and the left bron- × 2.6  cm) located at the tracheal bifurcation. It has chus, and the AVA tightly adhered to the esophageal wall. smooth borders with no obvious nodules. No other aneu- We used an electrocoagulation hook and an ultrasonic rysmal malformations were detected. The esophagus was scalpel to open the mediastinal pleura at the junction of obviously compressed on the corresponding plane. At the the AVA and the superior vena cava, denuded the AV, same time, contrast-enhanced CT suggests unremarkable and cut it off with an ultrasonic scalpel after hemolock Fig. 1 A Chest contrast-enhanced CT revealed that the mass located at the tracheal bifurcation. B Bronchoscopy revealed external pressure stenosis at the bronchial origin of the left lower lobe. C Three-dimensional reconstruction showed that the mass is a hemangioma originating from the azygos vein arch. There were two arteries from the aorta running among the tumor body, the esophagus and the trachea. D The tumor body obviously extruded the esophagus and trachea Y ao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 3 of 9 Fig. 2 A exploration revealed that the AVA was located in the azygos vein arch. B the venous aneurysm was completely removed. C A 16F drainage tube was placed in the surgical incision after single-port thoracoscopic surgery. D Histopathology showed thinning of the AV wall and loss of the smooth muscle layer of the vascular wall clip was clamped. Then, the stump end was pulled to sep - discovered from trauma, and none were proven to arate the tumor from the left main bronchus and esopha- be caused by trauma. Summarizing the previously gus wall. The ultrasonic scalpel and electrocoagulation reported cases, most of the patients were asymptomatic hook were used to dissociate the surrounding tissue of or had their AVA discovered incidentally on physi- the AVA. The aortic branches behind the tumor were cal examination for other reasons. Among the com- carefully separated, the ultrasonic scalpel was cut off, and mon symptoms in patients, 13 patients had chest pain the venous aneurysm was completely removed (Fig.  2B). [12–15, 17, 18, 20, 22, 26, 35, 44, 48, 60], 10 patients After the operation, a 16-F drainage tube was placed had chest tightness and even dyspnea [3, 6, 19–21, 23, in the surgical incision (Fig.  2C). The operation lasted 25, 45], 4 patients had cough [3, 9, 43, 53], 3 patients approximately 60  min, and intraoperative blood loss had symptoms of dysphagia [19, 43, 59], and 3 patients’ was 20  ml. The patient recovered well after surgery. All AVAs were accidentally discovered in trauma [3, 11]. symptoms of dysphagia disappeared after the operation, Contrast-enhanced CT and magnetic resonance imag- and the patient was discharged 3  days after the opera- ing (MRI) are the main noninvasive methods for the tion. Histopathology showed that the venous layer to be diagnosis of AVA. CT was completed in 72 patients affected is the media. Thinning of the AV wall and loss of of all reported 74 patients, and 22 patients completed the smooth muscle layer of the vascular wall. No malig- MRI. Further completion of gastroscopy and fiberoptic nant cells were seen. So it was considered an idiopathic bronchoscopy is an important supplement to CT and AVA (Fig. 2D). At the 1-month follow-up, CT showed no MRI when patients have symptoms such as dysphagia recurrence. and dyspnea [6, 60]. However, poor tumor enhance- ment or intratumoral thrombus formation in the AVA Discussion may lead to misdiagnosis of AVA as a solid tumor or AVA is a rare disease. AVA is generally defined as a mediastinal lymphadenopathy [24]. When a mediasti- diameter of the AV exceeding 2.5 times the normal nal mass does not exclude the diagnosis of azygosmo- diameter. According to its morphology, it is currently sis, needle biopsy is not recommended because of the divided into fusiform AVAs, with the overall expansion risk of massive bleeding [25, 26]. In the present case, of the AV, and saccular AVAs, with local expansion of the three-dimensional reconstruction of the patient the azygos wall [3]. According to the etiology, it can showed that the tumor was located in the AV, and be divided into (1) idiopathic AVA, (2) acquired AVA, the tumor was connected with the superior vena cava and (3) traumatic AVA [1]. As far as we know, a total of and the AV, which confirmed the diagnosis of AVA. 73 patients with AVAs have been reported in the avail- At the same time, the three-dimensional reconstruc- able literature, who are summarized in Table  1 [3–63]. tion showed the surrounding tissues of the tumor. The Among them, there were 29 males and 41 females. tumor was closely related to the esophagus and airway. Their ages ranged from 3  months to 86  years (mean, There were two arteries from the aorta running among 56.2 years), and their tumor diameters ranged from 1.2 the tumor body, the esophagus and trachea. During the to 15  cm (mean, 4.4  cm). There were 14 cases of fusi - operation, it was found that the tumor and esophagus form AVAs and 32 cases of saccular AVAs. There were were tightly adhered. When separating the posterior 54 cases of idiopathic AVAs and 6 cases of acquired part of the tumor, we predict the location of arterial AVAs. However, 3 patients’ AVAs was accidentally branches in advance. A combination of an ultrasonic Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 4 of 9 Table 1 Reported cases of Azygos Vein Aneurysm Study Age (y) Sex Symptom Origin AVA Type Measuring (cm) Diagnosis Associated diseases Treatment References Ko 33 M Chest tightness Idiopathic AVA Saccular 6.5 × 6.0 CXR, CT, MRI None Surgical resection [3] Ko 72 F Chest tightness Idiopathic AVA Saccular 6.0 × 5.0 CXR, CT, MRI None Surgical resection [3] Ko 55 M Arteriovenous fifistula Idiopathic AVA Saccular 3.7 × 3.0 CXR, CT End-stage renal disease Surgical resection [3] infection Ko 66 F Dyspnea, pulmonary Idiopathic AVA Saccular 2.8 × 2.4 CXR, CT After breast cancer Surgical resection [3] thromboembolism operation Ko 77 F Trauma Idiopathic AVA Fusiform 5.5 × 3.0 CXR, CT Diabetes mellitus Conservative [3] Ko 56 M Trauma Idiopathic AVA Fusiform 3.2 × 2.7 CXR, CT None Conservative [3] Ko 46 F Hemoptysis, lung Idiopathic AVA Fusiform 2.7 × 2.2 CXR, CT After rectal cancer VATS resection, left [3] metastasis operation upper lobectomy Ko 67 F Stroke Idiopathic AVA Fusiform 3.0 × 2.7 CXR, CT None Conservative [3] Ko 37 F Cough, hemoptysis Idiopathic AVA Fusiform 3.5 × 3.0 CXR, CT, MRI Bronchopulmonary VATS resection, right [3] sequestration lower lobectomy Ko 34 M Fever, cough Idiopathic AVA Fusiform 4.5 × 2.5 CXR, CT, MRI Drug addict, mediastinal Conservative [3] hemangiomatosis Choo 79 F Lower leg weakness Idiopathic AVA N/A 3.8 CXR, CT, MRI Vertebral disk disorder Conservative [4] He 41 F N/A Idiopathic AVA N/A N/A CXR, CT, MRI, phlebog- N/A Surgical resection [5] raphy Briones-Claudett 86 F Dysphagia, paresthesia N/A N/A N/A CXR, CT, Bronchoscopy, Aspergillus fumigatus Conservative [6] three-dimensional infection reconstruction Irurzun N/A N/A Cough, wheezing, Idiopathic AVA N/A N/A N/A N/A Transcatheter emboliza- [7] hiccups tion Figueiredo 78 M Depressed level of Acquired AVA Saccular 1.2 MRI Portal hypertension Conservative [8] consciousness Guo 28 M Cough, fever Acquired AVA Saccular N/A CT, three-dimensional After aneurysm resec- N/A [9] reconstruction tion Ichiki 76 M None Idiopathic AVA N/A 3.5 CXR, CT, MRI None VATS resection [10] Mohajeri 45 M Trauma Idiopathic AVA Saccular 3.8 × 2.8 CXR, CT, MRI None Conservative [11] DeMaio 28 F Chest pain, neurologic Acquired AVA Fusiform N/A CT, phlebography A motor vehicle collision Stent graft implantation [12] deficits, shock Weber 15 M Chest pain Idiopathic AVA Saccular 2.6 × 4.0 CXR, CT, MRI, PET Pulmonary embolus Transcatheter emboliza- [13] tion Favelier 78 F Chest pain Idiopathic AVA N/A 6.0 × 5.0 MRI, phlebography Pulmonary embolus Stent graft implantation [14] Icard 68 M Chest pain Idiopathic AVA Saccular 6.0 CXR, CT None Surgical resection [15] Tang 42 M None Idiopathic AVA Saccular 4.2 × 6.7 × 4.0 CT, three-dimensional N/A Surgical resection [16] reconstruction Du 70 F Chest pain Idiopathic AVA Saccular 4.0 × 3.5 × 3.4 CT, phlebography None VATS resection [17] Du 53 M None Idiopathic AVA Saccular 5.5 × 3.5 × 4.0 CT None Conservative [17] Y ao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 5 of 9 Table 1 (continued) Study Age (y) Sex Symptom Origin AVA Type Measuring (cm) Diagnosis Associated diseases Treatment References Du 60 F Cough N/A Saccular 2.1 × 2.0 × 2.0 CT None Oral anticoagulation [17] Xie 70 F Chest pain N/A Fusiform 4.0 × 3.5 × 3.4 CT None VATS resection [18] Morton 73 F Dysphagia, odynopha- Idiopathic AVA Saccular 5.5 × 5.0 CT, phlebography Chronic obstructive Transcatheter coil [19] gia, chest tightness pulmonary disease, embolization gastroesophageal reflux disease, diverticulitis, osteochondroma of the right scapula after surgery Wang 53 F Chest tightness, choking Idiopathic AVA Saccular 4.2 × 3.7 × 2.6 CT None VATS resection [20] Choi 0.25 M Chest tightness Idiopathic AVA Saccular 10 CXR, CT, T TE Pulmonary embolus Surgical resection [21] Takamori 43 N/A None Idiopathic AVA Saccular 4.7 × 3.5 × 2.4 CT None VATS resection [22] Zhang 36 M Chest pain, Chest tight- N/A Fusiform 5.5 × 4.0 × 4.2 CT None Surgical resection [23] ness Gomez N/A N/A N/A Idiopathic AVA N/A N/A CT, phlebography N/A N/A [24] Rothman 26 M Dachycardia, Chest Idiopathic AVA Fusiform 5 × 2.5 × 2.5 CXR, CT N/A Stent graft implantation [25] tightness Hatachi 56 F Chest pain Idiopathic AVA Fusiform 4.3 × 3.6 CT None VATS resection [26] Kurihara 73 F None Idiopathic AVA N/A 3.5 × 3.0 × 2.5 CXR, CT None Surgical resection [27] Zhang 42 M None Idiopathic AVA Saccular 5.0 CT None Surgical resection [28] Ueda 60 F None Idiopathic AVA N/A 2.5 CXR, CT None VATS resection [29] Lee 38 F None Idiopathic AVA Fusiform 3.0 CXR, CT None VATS resection [30] Miura 79 F None Idiopathic AVA Fusiform 6.6 × 6.5 × 4.5 CXR, CT, MRI None VATS resection [31] Guo 49 F None N/A Saccular 5.0 × 4.0 × 3.0 CT None Robot-assisted VATS [32] resection Hu 23 M None Acquired AVA Saccular 5.4 × 4.5 × 4.0 CT, three-dimensional ArteriovenousAneurysm VATS resection [33] reconstruction Obeso 74 F None N/A N/A 1.9 × 1.1 CT Lung cance VATS resection, right [34] upper lobectomy Davis 70 M Chest pain Idiopathic AVA Saccular 3.3 × 2.6 CT, phlebography Pulmonary embolism Stent graft implantation [35] Suzuki 79 M Pleural effusions Acquired AVA N/A 5.0 CXR, CT Mitral and tricuspid Surgical resection [36] valve regurgitation Córdoba 68 M Syncope Idiopathic AVA Saccular 3.8 CXR, CT, MRI Accessory fissure N/A [37] Seo 72 F Chest discomfort Idiopathic AVA N/A 3.5 CXR, MRI, PET None N/A [38] Imori 62 F None Idiopathic AVA N/A 2.0 × 1.5 CT, EUS Lung cancer Conservative [39] Yang 75 F General weakness, Idiopathic AVA N/A 4.1 × 2.5 × 3.0 CXR, CT Cerebral infarction Oral anticoagulation [40] neurologic deficits Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 6 of 9 Table 1 (continued) Study Age (y) Sex Symptom Origin AVA Type Measuring (cm) Diagnosis Associated diseases Treatment References Ishikura 51 F None Idiopathic AVA N/A 6.0 CXR, CT None Surgical resection [41] Ichihara 64 M Hemoptysis Acquired AVA N/A N/A N/A Dilated submucosal N/A [42] bronchial vessels Gnanamuthu 73 M Cough and mild dys- Idiopathic AVA N/A 5.0 CXR, CT, PET Chronic obstructive Surgical resection [43] phagia airways disease Nakamura 37 F Chest pain and palpita- Idiopathic AVA Saccular 11.0 × 9.0 CXR, CT Pulmonary embolus Embolectomy [44] tions D’Souza 29 F Dyspnea Idiopathic AVA N/A 5.0 CXR, CR, MRI Ehlers-Danlos syndrome Stent graft implantation [45] type IV Abad 49 F None Idiopathic AVA N/A 3.0 × 3.5 × 1.5 CXR, CT, MRI None Surgical resection [46] Person 61 F Lightheadedness, nau- Idiopathic AVA N/A N/A CXR, CT Hypothyroidism VATS resection [47] sea, and leg weakness Dilege 72 F Chest pain N/A N/A N/A CXR, CT, MRI N/A Surgical resection [48] Bobbio 66 F None N/A N/A N/A CXR, CT Esophageal carcinoma Surgical resection [49] Sakaguchi 52 M None Idiopathic AVA Saccular 4.0 × 3.0 × 3.0 CXR, CT, MRI None Surgical resection [50] Gallego 64 F Cough, wheezing Idiopathic AVA N/A 3.5 CXR, CT, MRI None N/A [51] Poll 46 F None Idiopathic AVA N/A 8.0 CXR, CT, MRI Sickle cell anemia Conservative [52] Watanabe 64 F Cough, fever Idiopathic AVA Saccular 3.0 CXR, CT, MRI, TEE None Surgical resection [53] Mehta 70 F None N/A N/A N/A CXR, CT Rectal carcinoma N/A [54] Lena 70 M Hematoma involving Idiopathic AVA N/A 3.0 × 3.0 CXR, CT, TEE Lung cancer, coronary N/A [55] the pectoralis major heart disease Kurihara 62 M N/A N/A Saccular N/A N/A N/A N/A [56] Seebauer 54 F Superior vena cava Idiopathic AVA Saccular 15.0 × 7.0 CXR, CT, TEE, phlebog- None Surgical resection [57] occlusion syndrome raphy Ikushima N/A N/A N/A N/A N/A N/A 4D-flow MRI N/A VATS resection [58] Bhojwani 17 M Dysphagia, postprandial Idiopathic AVA N/A N/A CT Heterotaxy syndrome Oesophagostomy [59] emesis Savu 74 F Chest pain N/A Saccular 3 × 4 CXR, CT, bronchoscopy Heart failure Surgical resection [60] Zhou 65 M None Idiopathic AVA Saccular 2.6 × 2.0 × 2.7 CT N/A VATS resection [61] Zhou 71 F Dizziness Idiopathic AVA Saccular 3.8 × 3.2 × 3.9 CT Hypertension Conservative [61] Zhang 46 F None N/A Saccular 5.0 CT None VATS resection [62] Sun 52 M None N/A Saccular 4.4 × 3.4 CT None VATS resection [63] This case 56 M Dysphagia Idiopathic AVA Fusiform 8.1 × 7.5 CT, bronchoscopy, None VATS resection three-dimensional reconstruction F female, M male, N/A information not available, AVA Azygos vein aneurysms, CT Computed tomography, CXR chest radiography, MRI magnetic resonance imaging, TEE transesophageal echocardiography, PET positron emission tomography, VATS video-assisted thoracoscopic surgery Y ao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 7 of 9 scalpel and electrocoagulation hook was used to care- risks associated with surgery. Finally, the tumor was suc- fully separate the area of the aortic branch to avoid the cessfully resected safely under single-port thoracoscopic risk of bleeding. Therefore, preoperative three-dimen - surgery. We have proved that single-port thoracoscopic sional reconstruction plays an important role in the surgery is safe and effective for the treatment of AVA. diagnosis of AVAs and the formulation of surgical plans At the same time, interventional therapy is a relatively [6, 8, 16, 33]. new option for AVA. There are case reports of success - Currently, there are no guidelines on the optimal treat- ful implantation of covered stents within the aneurysm ment of AVAs. In a previous report, conservative obser- [12, 14, 25, 35], transcatheter embolization of the aneu- vation combined with oral anticoagulation was successful rysms [7, 13] and Amplatzer closure occlusion of tumor in asymptomatic AVA patients. Ko et  al. [3] reported blood vessels [35]. Although interventional treatment that only 2 of 10 patients with idiopathic AVAs under- has less trauma than surgery. However, patients should went surgical resection immediately after diagnosis. Six be treated with surgery in case of excessive physiologi- patients underwent surgery due to thrombosis and tumor cal curvature of the AV anticipates difficulty in passing enlargement during follow-up. Patients should be re- the interventional guidewire, compression of adjacent evaluated regularly, and surgery or interventional therapy structures, appearance of clinical symptoms, thrombus should strongly be considered in cases of compression formation in the aneurysm sac in patients, severe pul- of adjacent structures, appearance of clinical symptoms, monary embolism requiring surgical intervention. We thrombus formation in the aneurysm sac in patients with believe that the surgical resection, especially minimally oral anticoagulation, contraindication to oral anticoagu- invasive resection, is simpler and safer in patients with lation, pulmonary embolism, and considerable increase AVAs and therefore generally recommend earlier treat- in the diameter of the AVA [1, 3, 18, 20]. In 21 of 72 ment even if there is no thrombus within the AVA [1, patients, thoracotomy or thoracoscopic-assisted thora- 29]. At the same time, embolization cannot prevent cotomy was performed to remove the AVA. With the thrombus migration or tumor compression symptoms advancement of endoscopic techniques, 19 of 72 patients [13]. Moreover, the embolization device may aggravate were also reported to have AVAs removed by three- compression symptoms after thrombosis. port or four-port thoracoscopic surgery, and 1 AVA was resected by robotic-assisted four-port endoscopy [3–63]. Single-port thoracoscopy causes less surgical trauma and Conclusion pain. With the development of minimally invasive tech- In conclusion, some patients may have symptoms such niques, surgeon are increasingly selecting the single-port as dysphagia and chest tightness due to tumor com- thoracoscope instead of the multi-port thoracoscope in pression of the esophagus and trachea. Preoperative thoracic surgery. We finally decided to select single-port three-dimensional reconstruction greatly helps sur- thoracotomy AVA resection which had been no reports geons clarify the disease diagnoses, formulate surgical until this one. Although the single-port thoracoscope plans, avoid damage to the surrounding vital organs, has less space to operate and more interference between and reduce intraoperative bleed. We proved that mini- operating instruments, the surgeon can overcome that mally invasive single-port thoracoscopic surgery is also through constant practice. The single-port thoracoscope safe and effective for the treatment of AVA. also has its unique advantages. The single-port thora - coscope lens is in the same direction as the operator’s Abbreviations vision. So the operation direction is in the same as the AV Azygos vein vision on the display that making the operation more AVA Azygos vein aneurysm CT Computed tomography accurate. However, the multi-port thoracoscope lens MRI Magnetic resonance imaging forms an angle with the operator’s direction of opera- VATS Video-assisted thoracoscopic surgery tion. So there is a visual deviation between the direction Acknowledgements of operation and the display, which may increase diffi - Not applicable. culty and the risk of bleeding for vascular operation. Dur- ing the surgery, we avoided unnecessary clamping of the Author contributions Participated in the care of the patient: YY, XX, GL, XL, JL. Performed the mass to prevent bleeding. The proximal end of the AVA literature review and drafted the manuscript: YY, QH, YL, CL, YY, YW, and LL. was blocked first to prevent thrombus migration from Obtained the image data: YY, QH. Critical Review: JL, XX. All authors read and causing pulmonary embolism. At the same time, the sur- approved the final manuscript. gical plan was formulated based on preoperative three- Funding dimensional reconstruction. We predict the location of None. arterial branches in advance, which further reduces the Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 8 of 9 Availability of data and materials 20. 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J Minim Access Surg. 2021;17(3):395–8. 60. Savu C, Melinte A, Balescu I, et al. Azygos vein aneurysm mimicking a mediastinal mass. In Vivo. 2020;34(4):2135–40. 61. Zhou ZY, Shen HL, Du HD, et al. Idiopathic azygos vein aneurysm: a report of 2 cases and literature review. Anhui Med Pharm J. 2021;25(12):2463–5. 62. Zhang CX, Zhao S, Li XN, et al. A case report of thoracoscopic surgery for azygostomy. Chin J Gerontol. 2012;32(1):150–1. 63. Sun ZW, Liu GW, Wang SD, et al. Resection of an azygos vein aneurysm by video-assisted thoracic surgery. Chin J Thorac Cardiovasc Surg. 2017;33(1):55–55. Publisher’s Note Springer Nature remains neutral with regard to jurisdictional claims in pub- lished maps and institutional affiliations. Re Read ady y to to submit y submit your our re researc search h ? Choose BMC and benefit fr ? Choose BMC and benefit from om: : fast, convenient online submission thorough peer review by experienced researchers in your field rapid publication on acceptance support for research data, including large and complex data types • gold Open Access which fosters wider collaboration and increased citations maximum visibility for your research: over 100M website views per year At BMC, research is always in progress. Learn more biomedcentral.com/submissions http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Cardiothoracic Surgery Springer Journals

Single-port thoracoscopic removal of an azygos vein aneurysm: a case report and literature review

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10.1186/s13019-023-02143-2
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Abstract

Background Azygos vein aneurysms (AVAs) are extremely rare. The majority of patients have no obvious clinical symptoms, so they are found by physical examination or by chance. There is limited clinical treatment experience that can be referred to, and there are no clear guidelines or research evidence standardizing the surgical and interven- tional therapy. Here, we report a patient with idiopathic AVA whose three-dimensional reconstruction of the tumor was completed before surgery. On the basis of three-dimensional reconstruction, single-port thoracoscopic resection of the AVA was successfully completed and reported for the first time. The previously reported cases are summarized to provide guidance for the diagnosis and treatment of patients with AVAs. Case presentation A 56-year-old man was transferred to our hospital due to “dysphagia”. The diagnosis of AVA was made after enhanced computed tomography, gastroscopy, fiberoptic bronchoscopy, and three-dimensional recon- struction. Congenital weakness or degenerative changes causes the vein walls to be extremely thin that the AVA had the risk of ruptur. Furthermore, the patient had symptoms of dysphagia, he received single-port thoracoscopic surgery. After the operation, his dysphagia disappeared. The postoperative pathology confirmed hemangioma. The patient was discharged 3 days after surgery without any complications. Conclusions AVAs are rare. Preoperative three-dimensional reconstruction can greatly help surgeons clarify the disease diagnosis, formulate the surgical plan, avoid damage to the surrounding vital organs, and reduce intraopera- tive bleeding. Thoracoscopic surgery to remove AVAs is difficult and has a high risk of bleeding, while more minimally invasive single-port thoracoscopic surgery is also safe and effective for the treatment of AVAs. Keywords Azygos vein aneurysm, Hemangioma of the azygos vein arch, Single-port thoracoscopic surgery, Three- dimensional reconstruction, Case report Background Yi Yao and Qiuxia Hu contributed to this work equally and thus are co-first authors Azygos vein aneurysms (AVAs) are rare [1]. In radiologic *Correspondence: studies, the normal diameter of the AV is defifined as no Ji Li more than 1  cm [2]. Underlying causes of AVA forma- njyyxxwklcy@163.com 1 tion that have been proposed include cardiac decompen- Department of Cardiothoracic Surgery, The First People’s Hospital of Neijiang, No. 1866, West Section of Hanan Avenue, Shizhong District, sation [3, 4], portal hypertension [4, 5], pregnancy [3], Neijiang 641000, Sichuan, China and compression of the SVC due to neoplasms [3, 5]. It Department of Obstetrics and Gynecology, The First People’s Hospital may occur rupture, thromboembolism, mediastinal mass of Neijiang, No. 1866, West Section of Hanan Avenue, Shizhong District, Neijiang 641000, Sichuan, China effects, and pulmonary artery hypertension with progres - sive gradual enlargement of the AVA [3]. Because most © The Author(s) 2023. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. The Creative Commons Public Domain Dedication waiver (http:// creat iveco mmons. org/ publi cdoma in/ zero/1. 0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 2 of 9 patients are asymptomatic, the diagnosis is mostly an enhancement in the arterial phase. It showed delayed incidental finding. The optimal management of AVAs enhancement with a CT value of approximately 118 HU remains uncertain, and there is no clear distinction of in the venous phase (Fig.  1A). Bronchoscopy revealed criteria indicating conservative and surgical or interven- external pressure stenosis at the bronchial origin of the tional therapy. Usually, surgical resection is required in left lower lobe (Fig.  1B). Gastroscopy showed no obvi- cases of compression of adjacent structures, appearance ous abnormalities. The initial diagnosis was hemangioma of clinical symptoms, azygos vein (AV) thrombus for- or bronchial cyst. Three-dimensional reconstruction mation in patients with oral anticoagulation, contrain- showed that the mass was connected to the AV and supe- dication to oral anticoagulation, pulmonary embolism, rior vena cava. The mass body obviously extruded into and considerable increase in the diameter of an AVA the esophagus and trachea. There were two arteries from [1]. Due to the high difficulty of thoracoscopic surgi - the aorta running among the mass body, the esophagus cal resection and the risk of embolism migration. Most and the trachea (Fig.  1C, D). These findings revealed patients underwent thoracotomy to remove AVAs, and that the mass is considered to be initial diagnosed as a only a few cases reported successful thoracoscopic resec- hemangioma originating from the azygos vein arch. Sur- tion [1]. There is no report of single-port thoracoscopic gical resection should take care with the aortic branch resection of an AVA. On the basis of three-dimensional behind the AVA to prevent uncontrollable bleed. At the reconstruction, we report a case of single-port thora- same time, we need to protect the vagus nerve and the coscopic-based management of an idiopathic AVA with thoracic duct. Avoid damaging them to prevent gastroin- symptoms of dysphagia. At the same time, we review the testinal symptoms and chylothorax. symptoms, diagnosis, and treatment of AVAs in the pub- Dysphagia caused by AVA extruded the esophagus and lished literature. the left bronchus. Interventional treatment may can’t cure dysphagia symptom. In addition, interventional Case report treatment may not prevent future migration and embo- A 56-year-old man complained of dysphagia. He was lization of the thrombus. So, we chose single-port video- referred to our hospital without special treatment assisted thoracoscopic surgery (VATS) to respect the because of a mediastinal mass on chest computed tomog- AVA under double-lumen tracheal intubation 3 days after raphy (CT). The patient had no relevant medical history. hospitalization. The patient was in the left lateral decu - He denied any falls or other trauma. Body temperature bitus position during surgery. The intraoperative explora - was 37.0 °C, heart rate was 78 beats/min, blood pressure tion results were consistent with the three-dimensional was 127/74 mmHg, and respiratory rate was 20 breaths/ reconstruction. The AVA was located in the azygos vein min. Breath sounds were normal. No moist or dry rales. arch, and there was no abnormal pulmonary artery or No heart murmurs or muffled heart tones. Contrast- pulmonary vein connected with it (Fig.  2A). The AVA enhanced CT revealed a mediastinal mass (4.2 × 3.7 obviously extruded into the esophagus and the left bron- × 2.6  cm) located at the tracheal bifurcation. It has chus, and the AVA tightly adhered to the esophageal wall. smooth borders with no obvious nodules. No other aneu- We used an electrocoagulation hook and an ultrasonic rysmal malformations were detected. The esophagus was scalpel to open the mediastinal pleura at the junction of obviously compressed on the corresponding plane. At the the AVA and the superior vena cava, denuded the AV, same time, contrast-enhanced CT suggests unremarkable and cut it off with an ultrasonic scalpel after hemolock Fig. 1 A Chest contrast-enhanced CT revealed that the mass located at the tracheal bifurcation. B Bronchoscopy revealed external pressure stenosis at the bronchial origin of the left lower lobe. C Three-dimensional reconstruction showed that the mass is a hemangioma originating from the azygos vein arch. There were two arteries from the aorta running among the tumor body, the esophagus and the trachea. D The tumor body obviously extruded the esophagus and trachea Y ao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 3 of 9 Fig. 2 A exploration revealed that the AVA was located in the azygos vein arch. B the venous aneurysm was completely removed. C A 16F drainage tube was placed in the surgical incision after single-port thoracoscopic surgery. D Histopathology showed thinning of the AV wall and loss of the smooth muscle layer of the vascular wall clip was clamped. Then, the stump end was pulled to sep - discovered from trauma, and none were proven to arate the tumor from the left main bronchus and esopha- be caused by trauma. Summarizing the previously gus wall. The ultrasonic scalpel and electrocoagulation reported cases, most of the patients were asymptomatic hook were used to dissociate the surrounding tissue of or had their AVA discovered incidentally on physi- the AVA. The aortic branches behind the tumor were cal examination for other reasons. Among the com- carefully separated, the ultrasonic scalpel was cut off, and mon symptoms in patients, 13 patients had chest pain the venous aneurysm was completely removed (Fig.  2B). [12–15, 17, 18, 20, 22, 26, 35, 44, 48, 60], 10 patients After the operation, a 16-F drainage tube was placed had chest tightness and even dyspnea [3, 6, 19–21, 23, in the surgical incision (Fig.  2C). The operation lasted 25, 45], 4 patients had cough [3, 9, 43, 53], 3 patients approximately 60  min, and intraoperative blood loss had symptoms of dysphagia [19, 43, 59], and 3 patients’ was 20  ml. The patient recovered well after surgery. All AVAs were accidentally discovered in trauma [3, 11]. symptoms of dysphagia disappeared after the operation, Contrast-enhanced CT and magnetic resonance imag- and the patient was discharged 3  days after the opera- ing (MRI) are the main noninvasive methods for the tion. Histopathology showed that the venous layer to be diagnosis of AVA. CT was completed in 72 patients affected is the media. Thinning of the AV wall and loss of of all reported 74 patients, and 22 patients completed the smooth muscle layer of the vascular wall. No malig- MRI. Further completion of gastroscopy and fiberoptic nant cells were seen. So it was considered an idiopathic bronchoscopy is an important supplement to CT and AVA (Fig. 2D). At the 1-month follow-up, CT showed no MRI when patients have symptoms such as dysphagia recurrence. and dyspnea [6, 60]. However, poor tumor enhance- ment or intratumoral thrombus formation in the AVA Discussion may lead to misdiagnosis of AVA as a solid tumor or AVA is a rare disease. AVA is generally defined as a mediastinal lymphadenopathy [24]. When a mediasti- diameter of the AV exceeding 2.5 times the normal nal mass does not exclude the diagnosis of azygosmo- diameter. According to its morphology, it is currently sis, needle biopsy is not recommended because of the divided into fusiform AVAs, with the overall expansion risk of massive bleeding [25, 26]. In the present case, of the AV, and saccular AVAs, with local expansion of the three-dimensional reconstruction of the patient the azygos wall [3]. According to the etiology, it can showed that the tumor was located in the AV, and be divided into (1) idiopathic AVA, (2) acquired AVA, the tumor was connected with the superior vena cava and (3) traumatic AVA [1]. As far as we know, a total of and the AV, which confirmed the diagnosis of AVA. 73 patients with AVAs have been reported in the avail- At the same time, the three-dimensional reconstruc- able literature, who are summarized in Table  1 [3–63]. tion showed the surrounding tissues of the tumor. The Among them, there were 29 males and 41 females. tumor was closely related to the esophagus and airway. Their ages ranged from 3  months to 86  years (mean, There were two arteries from the aorta running among 56.2 years), and their tumor diameters ranged from 1.2 the tumor body, the esophagus and trachea. During the to 15  cm (mean, 4.4  cm). There were 14 cases of fusi - operation, it was found that the tumor and esophagus form AVAs and 32 cases of saccular AVAs. There were were tightly adhered. When separating the posterior 54 cases of idiopathic AVAs and 6 cases of acquired part of the tumor, we predict the location of arterial AVAs. However, 3 patients’ AVAs was accidentally branches in advance. A combination of an ultrasonic Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 4 of 9 Table 1 Reported cases of Azygos Vein Aneurysm Study Age (y) Sex Symptom Origin AVA Type Measuring (cm) Diagnosis Associated diseases Treatment References Ko 33 M Chest tightness Idiopathic AVA Saccular 6.5 × 6.0 CXR, CT, MRI None Surgical resection [3] Ko 72 F Chest tightness Idiopathic AVA Saccular 6.0 × 5.0 CXR, CT, MRI None Surgical resection [3] Ko 55 M Arteriovenous fifistula Idiopathic AVA Saccular 3.7 × 3.0 CXR, CT End-stage renal disease Surgical resection [3] infection Ko 66 F Dyspnea, pulmonary Idiopathic AVA Saccular 2.8 × 2.4 CXR, CT After breast cancer Surgical resection [3] thromboembolism operation Ko 77 F Trauma Idiopathic AVA Fusiform 5.5 × 3.0 CXR, CT Diabetes mellitus Conservative [3] Ko 56 M Trauma Idiopathic AVA Fusiform 3.2 × 2.7 CXR, CT None Conservative [3] Ko 46 F Hemoptysis, lung Idiopathic AVA Fusiform 2.7 × 2.2 CXR, CT After rectal cancer VATS resection, left [3] metastasis operation upper lobectomy Ko 67 F Stroke Idiopathic AVA Fusiform 3.0 × 2.7 CXR, CT None Conservative [3] Ko 37 F Cough, hemoptysis Idiopathic AVA Fusiform 3.5 × 3.0 CXR, CT, MRI Bronchopulmonary VATS resection, right [3] sequestration lower lobectomy Ko 34 M Fever, cough Idiopathic AVA Fusiform 4.5 × 2.5 CXR, CT, MRI Drug addict, mediastinal Conservative [3] hemangiomatosis Choo 79 F Lower leg weakness Idiopathic AVA N/A 3.8 CXR, CT, MRI Vertebral disk disorder Conservative [4] He 41 F N/A Idiopathic AVA N/A N/A CXR, CT, MRI, phlebog- N/A Surgical resection [5] raphy Briones-Claudett 86 F Dysphagia, paresthesia N/A N/A N/A CXR, CT, Bronchoscopy, Aspergillus fumigatus Conservative [6] three-dimensional infection reconstruction Irurzun N/A N/A Cough, wheezing, Idiopathic AVA N/A N/A N/A N/A Transcatheter emboliza- [7] hiccups tion Figueiredo 78 M Depressed level of Acquired AVA Saccular 1.2 MRI Portal hypertension Conservative [8] consciousness Guo 28 M Cough, fever Acquired AVA Saccular N/A CT, three-dimensional After aneurysm resec- N/A [9] reconstruction tion Ichiki 76 M None Idiopathic AVA N/A 3.5 CXR, CT, MRI None VATS resection [10] Mohajeri 45 M Trauma Idiopathic AVA Saccular 3.8 × 2.8 CXR, CT, MRI None Conservative [11] DeMaio 28 F Chest pain, neurologic Acquired AVA Fusiform N/A CT, phlebography A motor vehicle collision Stent graft implantation [12] deficits, shock Weber 15 M Chest pain Idiopathic AVA Saccular 2.6 × 4.0 CXR, CT, MRI, PET Pulmonary embolus Transcatheter emboliza- [13] tion Favelier 78 F Chest pain Idiopathic AVA N/A 6.0 × 5.0 MRI, phlebography Pulmonary embolus Stent graft implantation [14] Icard 68 M Chest pain Idiopathic AVA Saccular 6.0 CXR, CT None Surgical resection [15] Tang 42 M None Idiopathic AVA Saccular 4.2 × 6.7 × 4.0 CT, three-dimensional N/A Surgical resection [16] reconstruction Du 70 F Chest pain Idiopathic AVA Saccular 4.0 × 3.5 × 3.4 CT, phlebography None VATS resection [17] Du 53 M None Idiopathic AVA Saccular 5.5 × 3.5 × 4.0 CT None Conservative [17] Y ao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 5 of 9 Table 1 (continued) Study Age (y) Sex Symptom Origin AVA Type Measuring (cm) Diagnosis Associated diseases Treatment References Du 60 F Cough N/A Saccular 2.1 × 2.0 × 2.0 CT None Oral anticoagulation [17] Xie 70 F Chest pain N/A Fusiform 4.0 × 3.5 × 3.4 CT None VATS resection [18] Morton 73 F Dysphagia, odynopha- Idiopathic AVA Saccular 5.5 × 5.0 CT, phlebography Chronic obstructive Transcatheter coil [19] gia, chest tightness pulmonary disease, embolization gastroesophageal reflux disease, diverticulitis, osteochondroma of the right scapula after surgery Wang 53 F Chest tightness, choking Idiopathic AVA Saccular 4.2 × 3.7 × 2.6 CT None VATS resection [20] Choi 0.25 M Chest tightness Idiopathic AVA Saccular 10 CXR, CT, T TE Pulmonary embolus Surgical resection [21] Takamori 43 N/A None Idiopathic AVA Saccular 4.7 × 3.5 × 2.4 CT None VATS resection [22] Zhang 36 M Chest pain, Chest tight- N/A Fusiform 5.5 × 4.0 × 4.2 CT None Surgical resection [23] ness Gomez N/A N/A N/A Idiopathic AVA N/A N/A CT, phlebography N/A N/A [24] Rothman 26 M Dachycardia, Chest Idiopathic AVA Fusiform 5 × 2.5 × 2.5 CXR, CT N/A Stent graft implantation [25] tightness Hatachi 56 F Chest pain Idiopathic AVA Fusiform 4.3 × 3.6 CT None VATS resection [26] Kurihara 73 F None Idiopathic AVA N/A 3.5 × 3.0 × 2.5 CXR, CT None Surgical resection [27] Zhang 42 M None Idiopathic AVA Saccular 5.0 CT None Surgical resection [28] Ueda 60 F None Idiopathic AVA N/A 2.5 CXR, CT None VATS resection [29] Lee 38 F None Idiopathic AVA Fusiform 3.0 CXR, CT None VATS resection [30] Miura 79 F None Idiopathic AVA Fusiform 6.6 × 6.5 × 4.5 CXR, CT, MRI None VATS resection [31] Guo 49 F None N/A Saccular 5.0 × 4.0 × 3.0 CT None Robot-assisted VATS [32] resection Hu 23 M None Acquired AVA Saccular 5.4 × 4.5 × 4.0 CT, three-dimensional ArteriovenousAneurysm VATS resection [33] reconstruction Obeso 74 F None N/A N/A 1.9 × 1.1 CT Lung cance VATS resection, right [34] upper lobectomy Davis 70 M Chest pain Idiopathic AVA Saccular 3.3 × 2.6 CT, phlebography Pulmonary embolism Stent graft implantation [35] Suzuki 79 M Pleural effusions Acquired AVA N/A 5.0 CXR, CT Mitral and tricuspid Surgical resection [36] valve regurgitation Córdoba 68 M Syncope Idiopathic AVA Saccular 3.8 CXR, CT, MRI Accessory fissure N/A [37] Seo 72 F Chest discomfort Idiopathic AVA N/A 3.5 CXR, MRI, PET None N/A [38] Imori 62 F None Idiopathic AVA N/A 2.0 × 1.5 CT, EUS Lung cancer Conservative [39] Yang 75 F General weakness, Idiopathic AVA N/A 4.1 × 2.5 × 3.0 CXR, CT Cerebral infarction Oral anticoagulation [40] neurologic deficits Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 6 of 9 Table 1 (continued) Study Age (y) Sex Symptom Origin AVA Type Measuring (cm) Diagnosis Associated diseases Treatment References Ishikura 51 F None Idiopathic AVA N/A 6.0 CXR, CT None Surgical resection [41] Ichihara 64 M Hemoptysis Acquired AVA N/A N/A N/A Dilated submucosal N/A [42] bronchial vessels Gnanamuthu 73 M Cough and mild dys- Idiopathic AVA N/A 5.0 CXR, CT, PET Chronic obstructive Surgical resection [43] phagia airways disease Nakamura 37 F Chest pain and palpita- Idiopathic AVA Saccular 11.0 × 9.0 CXR, CT Pulmonary embolus Embolectomy [44] tions D’Souza 29 F Dyspnea Idiopathic AVA N/A 5.0 CXR, CR, MRI Ehlers-Danlos syndrome Stent graft implantation [45] type IV Abad 49 F None Idiopathic AVA N/A 3.0 × 3.5 × 1.5 CXR, CT, MRI None Surgical resection [46] Person 61 F Lightheadedness, nau- Idiopathic AVA N/A N/A CXR, CT Hypothyroidism VATS resection [47] sea, and leg weakness Dilege 72 F Chest pain N/A N/A N/A CXR, CT, MRI N/A Surgical resection [48] Bobbio 66 F None N/A N/A N/A CXR, CT Esophageal carcinoma Surgical resection [49] Sakaguchi 52 M None Idiopathic AVA Saccular 4.0 × 3.0 × 3.0 CXR, CT, MRI None Surgical resection [50] Gallego 64 F Cough, wheezing Idiopathic AVA N/A 3.5 CXR, CT, MRI None N/A [51] Poll 46 F None Idiopathic AVA N/A 8.0 CXR, CT, MRI Sickle cell anemia Conservative [52] Watanabe 64 F Cough, fever Idiopathic AVA Saccular 3.0 CXR, CT, MRI, TEE None Surgical resection [53] Mehta 70 F None N/A N/A N/A CXR, CT Rectal carcinoma N/A [54] Lena 70 M Hematoma involving Idiopathic AVA N/A 3.0 × 3.0 CXR, CT, TEE Lung cancer, coronary N/A [55] the pectoralis major heart disease Kurihara 62 M N/A N/A Saccular N/A N/A N/A N/A [56] Seebauer 54 F Superior vena cava Idiopathic AVA Saccular 15.0 × 7.0 CXR, CT, TEE, phlebog- None Surgical resection [57] occlusion syndrome raphy Ikushima N/A N/A N/A N/A N/A N/A 4D-flow MRI N/A VATS resection [58] Bhojwani 17 M Dysphagia, postprandial Idiopathic AVA N/A N/A CT Heterotaxy syndrome Oesophagostomy [59] emesis Savu 74 F Chest pain N/A Saccular 3 × 4 CXR, CT, bronchoscopy Heart failure Surgical resection [60] Zhou 65 M None Idiopathic AVA Saccular 2.6 × 2.0 × 2.7 CT N/A VATS resection [61] Zhou 71 F Dizziness Idiopathic AVA Saccular 3.8 × 3.2 × 3.9 CT Hypertension Conservative [61] Zhang 46 F None N/A Saccular 5.0 CT None VATS resection [62] Sun 52 M None N/A Saccular 4.4 × 3.4 CT None VATS resection [63] This case 56 M Dysphagia Idiopathic AVA Fusiform 8.1 × 7.5 CT, bronchoscopy, None VATS resection three-dimensional reconstruction F female, M male, N/A information not available, AVA Azygos vein aneurysms, CT Computed tomography, CXR chest radiography, MRI magnetic resonance imaging, TEE transesophageal echocardiography, PET positron emission tomography, VATS video-assisted thoracoscopic surgery Y ao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 7 of 9 scalpel and electrocoagulation hook was used to care- risks associated with surgery. Finally, the tumor was suc- fully separate the area of the aortic branch to avoid the cessfully resected safely under single-port thoracoscopic risk of bleeding. Therefore, preoperative three-dimen - surgery. We have proved that single-port thoracoscopic sional reconstruction plays an important role in the surgery is safe and effective for the treatment of AVA. diagnosis of AVAs and the formulation of surgical plans At the same time, interventional therapy is a relatively [6, 8, 16, 33]. new option for AVA. There are case reports of success - Currently, there are no guidelines on the optimal treat- ful implantation of covered stents within the aneurysm ment of AVAs. In a previous report, conservative obser- [12, 14, 25, 35], transcatheter embolization of the aneu- vation combined with oral anticoagulation was successful rysms [7, 13] and Amplatzer closure occlusion of tumor in asymptomatic AVA patients. Ko et  al. [3] reported blood vessels [35]. Although interventional treatment that only 2 of 10 patients with idiopathic AVAs under- has less trauma than surgery. However, patients should went surgical resection immediately after diagnosis. Six be treated with surgery in case of excessive physiologi- patients underwent surgery due to thrombosis and tumor cal curvature of the AV anticipates difficulty in passing enlargement during follow-up. Patients should be re- the interventional guidewire, compression of adjacent evaluated regularly, and surgery or interventional therapy structures, appearance of clinical symptoms, thrombus should strongly be considered in cases of compression formation in the aneurysm sac in patients, severe pul- of adjacent structures, appearance of clinical symptoms, monary embolism requiring surgical intervention. We thrombus formation in the aneurysm sac in patients with believe that the surgical resection, especially minimally oral anticoagulation, contraindication to oral anticoagu- invasive resection, is simpler and safer in patients with lation, pulmonary embolism, and considerable increase AVAs and therefore generally recommend earlier treat- in the diameter of the AVA [1, 3, 18, 20]. In 21 of 72 ment even if there is no thrombus within the AVA [1, patients, thoracotomy or thoracoscopic-assisted thora- 29]. At the same time, embolization cannot prevent cotomy was performed to remove the AVA. With the thrombus migration or tumor compression symptoms advancement of endoscopic techniques, 19 of 72 patients [13]. Moreover, the embolization device may aggravate were also reported to have AVAs removed by three- compression symptoms after thrombosis. port or four-port thoracoscopic surgery, and 1 AVA was resected by robotic-assisted four-port endoscopy [3–63]. Single-port thoracoscopy causes less surgical trauma and Conclusion pain. With the development of minimally invasive tech- In conclusion, some patients may have symptoms such niques, surgeon are increasingly selecting the single-port as dysphagia and chest tightness due to tumor com- thoracoscope instead of the multi-port thoracoscope in pression of the esophagus and trachea. Preoperative thoracic surgery. We finally decided to select single-port three-dimensional reconstruction greatly helps sur- thoracotomy AVA resection which had been no reports geons clarify the disease diagnoses, formulate surgical until this one. Although the single-port thoracoscope plans, avoid damage to the surrounding vital organs, has less space to operate and more interference between and reduce intraoperative bleed. We proved that mini- operating instruments, the surgeon can overcome that mally invasive single-port thoracoscopic surgery is also through constant practice. The single-port thoracoscope safe and effective for the treatment of AVA. also has its unique advantages. The single-port thora - coscope lens is in the same direction as the operator’s Abbreviations vision. So the operation direction is in the same as the AV Azygos vein vision on the display that making the operation more AVA Azygos vein aneurysm CT Computed tomography accurate. However, the multi-port thoracoscope lens MRI Magnetic resonance imaging forms an angle with the operator’s direction of opera- VATS Video-assisted thoracoscopic surgery tion. So there is a visual deviation between the direction Acknowledgements of operation and the display, which may increase diffi - Not applicable. culty and the risk of bleeding for vascular operation. Dur- ing the surgery, we avoided unnecessary clamping of the Author contributions Participated in the care of the patient: YY, XX, GL, XL, JL. Performed the mass to prevent bleeding. The proximal end of the AVA literature review and drafted the manuscript: YY, QH, YL, CL, YY, YW, and LL. was blocked first to prevent thrombus migration from Obtained the image data: YY, QH. Critical Review: JL, XX. All authors read and causing pulmonary embolism. At the same time, the sur- approved the final manuscript. gical plan was formulated based on preoperative three- Funding dimensional reconstruction. We predict the location of None. arterial branches in advance, which further reduces the Yao et al. Journal of Cardiothoracic Surgery (2023) 18:29 Page 8 of 9 Availability of data and materials 20. 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Journal

Journal of Cardiothoracic SurgerySpringer Journals

Published: Jan 16, 2023

Keywords: Azygos vein aneurysm; Hemangioma of the azygos vein arch; Single-port thoracoscopic surgery; Three-dimensional reconstruction; Case report

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