Case Study

Surgical intervention without lung resection for Pryce type I sequestration

Asian Cardiovascular & Thoracic Annals 2015, Vol. 23(7) 872–874 ß The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492315574389 aan.sagepub.com

Takeshi Kawaguchi, Takashi Tojo, Motoaki Yasukawa, Takashi Watanabe, Norikazu Kawai and Shigeki Taniguchi

Abstract Anomalous systemic arterial supply to normal basal segments of the lower lobe is a rare congenital anomaly. Resection of the affected lung with ligation of the anomalous artery is commonly performed in these patients. We report a case of this anomaly treated surgically with interruption of the anomalous artery after placing a Hem-o-Lok vascular clip. Interruption of the anomalous artery using this vascular clip was easy and safe, and the thoracoscopic approach was minimally invasive. The patient’s recovery was uneventful and he was doing well without recurrence 4 years after the operation.

Keywords Bronchopulmonary sequestration, pulmonary artery, respiratory system abnormalities, surgical instruments, thoracic surgery, video-assisted

Introduction Anomalous systemic arterial supply to normal basal segments of the lower lobe is a rare congenital anomaly known as Pryce type I sequestration. Resection of the affected lung with ligation of the anomalous artery is commonly performed in these patients.1,2 Lung-conserving surgical methods, such as reimplantation of the anomalous artery to the pulmonary artery or simple division of the anomalous artery, have rarely been reported.1,3,4 Transarterial embolization of an anomalous artery was recently reported to be a less invasive method.5 We describe a case of this anomaly that was surgically treated with interruption of the anomalous artery using a Hem-o-Lok clip (Weck Teleflex Medical, Durham, NC, USA).

Case report A 25-year-old man presented to our institution with recurrent hemoptysis. Contrast-enhanced chest computed tomography showed an anomalous systemic artery from the descending aorta to the left basal segments of the lung (Figure 1a). In addition, increased parenchymal density and dilated pulmonary vessels were seen in the affected segments (Figure 1b).

The pulmonary bronchi and veins were anatomically normal, and the pulmonary arteries supplying the basal segments could not be seen. A diagnosis of anomalous systemic arterial supply to the normal basal segments of the lower lobe was made on the basis of these computed tomography findings, and the patient was referred to our department for surgical intervention. Video-assisted thoracoscopic surgery was performed with 3 ports. The surfaces of the left basal segments were telangiectatic (Figure 2a); however, the superior segment of the lower lobe (S6) and the upper lobe were normal in appearance. The anomalous artery was easily identified just below the inferior pulmonary vein in the pulmonary ligament, and was minimally dissected and clamped with a Hem-o-Lok clip (Figure 2b). After clamping the anomalous artery, the telangiectatic changes in the basal segments improved. We waited Department of Thoracic and Cardiovascular Surgery, Nara Medical University School of Medicine, Nara, Japan Corresponding author: Takeshi Kawaguchi, Department of Thoracic and Cardiovascular Surgery, Nara Medical University School of Medicine, 840 Shijo-cho, Kashihara, Nara 634-8522, Japan. Email: [email protected]

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Figure 1. Preoperative contrast-enhanced chest computed tomography showing (a) an anomalous systemic artery from the descending aorta as well as (b) increased parenchymal density and dilated pulmonary vessels. (c) Recent unenhanced chest computed tomography showing the healthy affected segment 4 years after the operation.

Figure 2. Intraoperative macroscopic findings: (a) the surfaces of the left basal segments are noted to be telangiectatic, and (b) the anomalous artery clamped with a Hem-o-Lok clip.

for approximately 30 min to ensure an unremarkable appearance of the affected segments as well as to monitor hemodynamics and blood gases. Ultimately, we decided to complete the operation without resection of the affected basal segments. The patient’s recovery was uneventful, and he left the hospital on the 7th

postoperative day. After the operation, the increased parenchymal density and dilated pulmonary vessels of the left basal segments had improved on computed tomography (Figure 1c), and the patient’s hemoptysis had completely resolved. He was doing well without recurrence 4 years after the operation.

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Discussion Lung resection with ligation of the anomalous artery is commonly performed in patients with a systemic arterial supply to the normal basal segments of the lower lobe.1,2 Lung resection is preferred because pathological pulmonary hypertensive changes have already been established, and improvement of pulmonary function with reimplantation of the anomalous artery to the pulmonary artery is not expected.1 Interruption of flow through the anomalous artery without lung resection, i.e. surgical division of the anomalous artery or transarterial embolization, has been reported as an alternative method.3–5 The appropriateness of leaving the diseased lung segments is unknown. A lung-conserving procedure might be feasible in patients with an affected lung that has normal pulmonary artery branches.1 However, the presence of a normal pulmonary artery to diseased basal segments is difficult to determine even by pulmonary arteriography.3,4 Successful interruption of an anomalous artery using transarterial embolization has been reported in patients without normal pulmonary artery branches to the basal segments.5 In our case, the pulmonary arteries supplying the basal segments could not be detected on contrast-enhanced computed tomography. We did not perform catheter angiography because we did not think it would contribute to the surgical decision making. Preoperatively, we could not be certain that a lung-conserving procedure would be appropriate for this patient. After clamping the anomalous artery during the operation, the telangiectatic changes of the basal segments improved immediately. On confirming the absence of pulmonary ischemic complications, we decided to leave the basal segments intact. Although the criteria for conservation of the diseased segments are unknown, the macroscopic appearance of the segments and observation of intraoperative hemodynamic changes should be considered. Surgical approaches have a clear advantage over transarterial embolization from this perspective. In addition, a video-assisted thoracoscopic surgery approach is feasible and minimally invasive for this procedure. In this case, interruption of the anomalous artery was performed using a Hem-o-Lok clip. In previous reports, the artery was interrupted by simple ligation or stapling.1–4 The procedure using a Hem-o-Lok clip was easy and required minimal dissection of the anomalous artery, less than using an endoscopic-stapler.

The removability of the clip was also advantage in this case because its application to an anomalous artery was the first experience for us; if it did not work well, we could remove the clip. Hem-o-Lok clips are often applied during endoscopic surgeries.6 Although their application in patients with this congenital anomaly has not been reported, this vascular clip was useful for interrupting the anomalous artery. The patient has been doing well and has had no recurrence in the 4 years since the operation. Although evaluation of the long-term outcome is necessary, surgical interruption of the anomalous artery is feasible in patients with anomalous systemic arterial supply to normal basal segments of the lower lobe. In addition, a Hem-o-Lok clip was useful for interrupting the anomalous artery via video-assisted thoracoscopic surgery. Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

Conflict of interest statement None declared.

References 1. Yamanaka A, Hirai T, Fujimoto T, Hase M, Noguchi M and Konishi F. Anomalous systemic arterial supply to normal basal segment of the left lower lobe. Ann Thorac Surg 1999; 68: 332–338. 2. Mori S, Odaka M, Asano H, et al. Anomalous systemic arterial supply to the basal segment of the lung: feasible thoracoscopic surgery. Ann Thorac Surg 2013; 96: 990–994. 3. Baek WK, Cho J, Kim JT, et al. Systemic arterial supply to normal basal segment of the left lower lobe the along with pulmonary artery: is lung resection warranted? J Thorac Cardiovasc Surg 2006; 131: 742–743. 4. Barik R, Patnaik AN, Malempati AR, Nemani L. Pryce type I sequestration: no mosquito shooting. Asian Cardiovasc Thorac Ann 2014 Feb 4. [Epub ahead of print]. 5. Jiang S, Shi JY, Zhu XH, et al. Endovascular embolization of the complete type of anomalous systemic arterial supply to normal basal lung segments: a report of four cases and literature review. Chest 2011; 139: 1506–1513. 6. Ponsky L, Cherullo E, Moinzadeh A, et al. The Hemo-oLok clip is safe for laparoscopic nephrectomy: a multiinstitutional review. Urology 2008; 71: 593–596.

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Surgical intervention without lung resection for Pryce type I sequestration.

Anomalous systemic arterial supply to normal basal segments of the lower lobe is a rare congenital anomaly. Resection of the affected lung with ligati...
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