Ann Thorac Cardiovasc Surg 2017; 23: 31–35 

Case Report

Online June 16, 2016 doi: 10.5761/atcs.cr.16-00110

Partial Anomalous Pulmonary Venous Connection Coexisting with Lung Cancer: A Case Report and Review of Relevant Cases from the Literature Hidenori Kawasaki, MD, PhD,1 Yasuji Oshiro, MD, PhD,2 Naohiro Taira, MD,1 Tomonori Furugen, MD,1 Takaharu Ichi, MD,1 Tomofumi Yohena, MD, PhD,1 and Tsutomu Kawabata, MD, PhD1

A 45-year-old man had an abnormal shadow in the right lung field on an annual screening chest X-ray. He was diagnosed with Stage IA (cT1bN0M0) lung cancer. Initially, we did not notice an anomalous vein on non-contrast computed tomography. However, we found that the right upper lobe bronchus branched from the lateral wall of the right main bronchial orifice, above the level of the common right upper lobe bronchus. Therefore, the bronchus was thought to be a tracheal bronchus. We carefully reevaluated the patient using three-dimensional computed tomography angiography. This technique showed that the anomalous right superior pulmonary vein drained into the azygos vein along the superior vena cava. These findings confirmed a partial anomalous pulmonary venous connection of the right upper lobe. We performed video-assisted thoracoscopic right upper lobectomy and mediastinal lymph node dissection for definitive treatment for lung cancer and partial anomalous pulmonary venous connection. No hemodynamic problems occurred in the postoperative course. Keywords:  partial anomalous pulmonary venous connection, lung cancer, three-dimensional computed tomography angiography

Introduction Partial anomalous pulmonary venous connection (PAPVC) is a comparatively uncommon congenital vascular anomaly. PAPVC is frequently associated with congenital heart disease, such as atrial septal defect. However, PAPVC that is incidentally found on computed tomography (CT) in adults without heart disease is uncommon, and Department of Surgery, National Hospital Organization, Okinawa National Hospital, Ginowan, Okinawa, Japan 2Department of Radiology, National Hospital Organization, Okinawa National Hospital, Ginowan, Okinawa, Japan 1

Received: April 26, 2016; Accepted: May 27, 2016 Corresponding author: Hidenori Kawasaki, MD, PhD. Department of Surgery, National Hospital Organization, Okinawa National Hospital, 3-20-14, Ganeko, Ginowan, Okinawa 901-2214, Japan Email: [email protected] ©2017 The Editorial Committee of Annals of Thoracic and Cardiovascular Surgery. All rights reserved.

Ann Thorac Cardiovasc Surg Vol. 23, No. 1 (2017)

generally clinically insignificant. We report here a case of lung cancer coexisting with asymptomatic PAPVC, which was diagnosed with preoperative contrast three-dimensional computed tomography angiography (3D-CTA).

Case Report A 45-year-old man, who never smoked, was referred to our hospital for evaluation of an abnormal shadow in the right upper lung field on an annual screening chest X-ray taken in January 2015 (Fig. 1A). His past and family histories were unremarkable. He likes to run, and he had finished a full marathon several times. A physical examination showed no abnormalities and no lymphadenopathy. There was no evidence of vascular shunt sounds on the chest and neck. Routine laboratory tests were within the normal range. A chest X-ray showed a slightly high-intensity shadow in the right upper lung field. A chest non-contrast CT scan showed an irregular, circumscribed, ill-defined, 31

Kawasaki H, et al. (A)

(B)

Fig. 1  (A) Chest X-ray shows an abnormal shadow in the right upper lung field. (B) Chest CT shows the RULB branching from the lateral wall of the right main bronchial orifice, as a tracheal bronchus. CT: computed tomography; RULB: right upper lobe bronchus

(A)

(B)

Fig. 2  (A) Frontal view of 3D-CTA showing PV1-3 draining into the azygos vein along the superior vena cava. (B) Back view of 3D-CTA showing the RULB branching from the lateral wall of the right main bronchial orifice, as a tracheal bronchus. The length of the RULB is approximately 3 cm, which is longer than the common variation. 3D-CTA: three-dimensional computed tomography angiography; RULB: right upper lobe bronchus

partly solid nodule in the right upper lobe, measuring 2.5 cm in diameter. There was no evidence of enlargement of the lymph nodes. We could not detect any distant metastasis by systemic examinations, including brain magnetic resonance imaging and 18F-fluorodeoxy-glucose positron emission tomography/CT. We planned bronchofiberscopy to confirm the histological diagnosis of lung tumor. On careful review of the patient’s non-contrast CT images before bronchofiberscopy, we observed that the right upper lobe bronchus 32

(RULB) was branching from the lateral wall of the right main bronchial orifice, above the level of the common RULB. Therefore, the bronchus was thought to be a tracheal bronchus (Fig. 1B). The length of the RULB was approximately 3 cm, which is longer than the common variation, and segmental bronchi of the right upper lobe had two branches. We also observed that the right upper lobe pulmonary vein (PV1-3) did not connect to the left atrium, and the right middle lobe pulmonary vein drained into the left atrium as normal. We performed a contrast CT Ann Thorac Cardiovasc Surg Vol. 23, No. 1 (2017)

Partial Anomalous Pulmonary Venous Connection with Lung Cancer (A)

(B)

Fig. 3  (A) Contrast CT showing PV1-3 draining into the azygos vein along the superior vena cava. (B) Thoracoscopic view of the pulmonary hilum after dissecting the pleura. A PAPVC was observed, as predicted by 3D-CTA. CT: computed tomography; PAPVC: partial anomalous pulmonary venous connection; 3D-CTA: three-dimensional computed tomography angiography

scan and 3D-CTA, which showed that the PV1-3 drained into the azygos vein along the superior vena cava, and it confirmed PAPVC of the right upper lobe (Figs. 2A–2B and 3A). Pathological examination of right transbronchial lung tumor biopsies confirmed the diagnosis of adenocarcinoma. The patient was diagnosed with Stage IA (cT1bN0M0) lung cancer coexisting with of PAPVC in the same lobe. Spirometry showed that the vital capacity was 5010 mL and forced expiratory volume in 1.0 s was 4100 mL. Electrocardiography showed normal cardiovascular activity, and his preoperative Qp/Qs ratio by Doppler echocardiography was only 1.08. Lung cancer was located in the right upper lobe, in the same location as PAPVC. Therefore, we planned to perform right upper lobectomy and mediastinal lymph node dissection for definitive treatment of lung cancer and PAPVC. We considered that no hemodynamic problems would occur during and after the operation. Under general anesthesia and double-lumen intubation, we performed video-assisted thoracoscopic approach using three ports. An anomalous vessel, PV1-3, was noted after dissecting the pleura, which drained into the azygos vein from the right upper lobe (Fig. 3B). The PV1-3 was divided using a stapler as the usual maneuver, and right upper lobe lobectomy and lymph node dissection were performed. The operative time was 2 h and 51 min and the amount of bleeding was 30 mL. The patient’s postoperative course was uneventful, and no hemodynamic problems Ann Thorac Cardiovasc Surg Vol. 23, No. 1 (2017)

occurred. The patient was followed up with no recurrence for 16 months.

Discussion PAPVC is a comparatively uncommon congenital vascular anomaly that is found in only 0.1%–0.2% of the adult population using CT scans.1,2) In this vascular anomaly, one or more pulmonary veins drain into a systemic vein, resulting in a left-to-right shunt. Anomalous right-sided pulmonary veins drain into the superior vena cava, inferior vena cava, right atrium, azygos vein, portal vein, or hepatic vein. Additionally, anomalous left-sided pulmonary veins can drain into the left brachiocephalic vein, coronary sinus, or hemiazygos vein.2,3) The patient’s clinical severity is determined by the degree of the left-to-right shunt. Patients with a small amount of shunts are usually asymptomatic and these tend to not be found during their lifetime. These anomalous veins are sometimes incidentally detected during investigation of another disease, while patients with large shunts present with dyspnea, palpitations, and chest pain. Our patient had no symptoms, and he finished a full marathon. His lung cancer was located in the right upper lobe in the same location as PAPVC. Lobectomy was the definitive treatment for lung cancer and PAPVC, and no hemodynamic problems occurred during and after the operation. However, if PAPVC is located in a different lobe to be preserved, major lung resection for conditions, such 33

Kawasaki H, et al.

Table 1  Summary of the location and surgical procedures for PAPVC with lung cancer in 18 cases Preoperative diagnosis of PAPVC (N) Yes (9)

Positional relationship of PAPVC and LC (N)

Location of PAPVC (N)

Surgical procedures for PAPVC

Surgical procedures for LC (N)

Outcome after OP

Same lobe (6)

RU (3), RL (1), LU (1)

Simultaneous resection Simultaneous resection

Lobectomy (5)

No problems

LUS (1)

No problems

LU (1)

No (9)

Ipsilateral different lobe (2) Contrarateral (1)

RU (1) LU (1) RU (1)

None Revascularization Revascularization

RLL (1) LLL (1) LP (1)

No problems No problems No problems

Same lobe (6)

RU (2), LU (4)

Simultaneous resection

Lobectomy (6)

No problems

Ipsilateral different lobe (2) Contrarateral (1)

RU (1) LU (1) LU (1)

None None None

RMLBL (1) LLL (1) RP (1)

No problems No problems Heart failure

PAPVC: partial anomalous pulmonary venous connection; N: number of patients; LC: lung cancer; OP: operation; RU: right upper lobe; RL: right lower lobe; LU: left upper lobe; LL: left lower lobe; LUS: left upper segmentectomy; RLL: right lower lobectomy; LLL: left lower lobectomy; LP: left pneumonectomy; RMLBL: right middle and lower bilobectomy: RP: right pneumonectomy

as lung cancer, could increase the volume of left-to-right shunt flow and cause right-sided heart failure.4) In the English and Japanese literature, there are 17 reported cases of PAPVC associated with lung cancer.4–20) Including our case and these previous reports, preoperative diagnosis of PAPVC was obtained in nine (50%) cases. The other nine cases were not diagnosed as PAPVC before pulmonary resection. One patient developed heart failure after right pneumonectomy for lung cancer with coexisting left-sided PAPVC.4) Fortunately, six cases of PAPVC were recognized in the same lobe as lung cancer, and two cases of PAPVC were recognized in ipsilateral different lobe during the operation, and the postoperative course of each cases were uneventful (Table 1). Similar to these reported series, some cases of PAPVC could be overlooked or undetected at the initial radiological diagnosis because of the rare association, low incidence of symptoms, and lack of knowledge of physicians. In our case, the RULB had a tracheal bronchus, with branching from the lateral wall of the right main bronchial orifice, above the level of the common RULB, and this was a bronchial anomaly. The length of the RULB was approximately 3 cm, which is longer than the common variation, and segmental bronchi of the right upper lobe had two branches. We did not notice the coexisting PAPVC in the first non-contrast CT image. However, based on the anomalous findings of the bronchus, we reevaluated our patient using contrast 3D-CTA, and diagnosed him with coexisting PAPVC. Recently, the quality of CT has greatly 34

advanced, and it is able to detect minor vessels or bronchial anomalies. Oshiro et al.1) reported that 42% (5/12) of cases of PAPVC were associated with major bronchial anomalies, such as tracheal bronchus and accessory cardiac bronchus. If we encounter a bronchial anomaly on preoperative CT, we need to consider the possibility of coexisting PAPVC.

Conclusion PAPVC is a rare congenital anomaly. Thoracic surgeons should be aware of the possibility of the presence of PAPVC if they plan to perform a major lung resection of not only the affected lobe in lung cancer, but also the preserved lobe before a major lung resection to prevent unexpected complications. The presence of bronchial anomalies could indicate coexisting PAPVC.

Consent Written informed consent was obtained from the patient for publication of this case report and accompanying images.

Acknowledgements We thank Masayuki Tawata, and Daisuke Tanaka, radiological technologists, in our hospital for reconstruction of 3D-CTA. Ann Thorac Cardiovasc Surg Vol. 23, No. 1 (2017)

Partial Anomalous Pulmonary Venous Connection with Lung Cancer

Disclosure Statement The authors declare that they have no competing interests.

References 1) Oshiro Y, Murayama S, Miyagi S, et al. Simultaneous occurrence of partial anomalous pulmonary venous return and major bronchial anomaly: computed tomography findings in 5 adult patients. J Comput Assist Tomogr 2009; 33: 535-9. 2) Ho ML, Bhalla S, Bierhals A, et al. MDCT of partial anomalous pulmonary venous return (PAPVR) in adults. J Thorac Imaging 2009; 24: 89-95. 3) Tourmousoglou C, Kalogeropoulou C, Koletsis E, et al. Right upper lobe partial anomalous pulmonary venous connection. Case Rep Vasc Med 2014; 2014: 249896. 4) Black MD, Shamji FM, Goldstein W, et al. Pulmonary resection and contralateral anomalous venous drainage: a lethal combination. Ann Thorac Surg 1992; 53: 689-91. 5) Takamori S, Hayashi A, Nagamatsu Y, et al. Left partial anomalous pulmonary venous connection found during a lobectomy for lung cancer: report of a case. Surg Today 1995; 25: 982-3. 6) Takei H, Suzuki K, Asamura H, et al. Successful pulmonary resection of lung cancer in a patient with partial anomalous pulmonary venous connection: report of a case. Surg Today 2002; 32: 899-901. 7) Akiyama H, Okada D, Enomoto Y, et al. A case of right partial anomalous pulmonary venous connection found during A lobectomy for lung cancer. Jpn J Lung Cancer 2003; 43: 711-4. (in Japanese with English abstract) 8) Miwa K, Takamori S, Hayashi A, et al. Incidental partial anomalous pulmonary venous connection in left lung cancer. Jpn J Thorac Cardiovasc Surg 2004; 52: 189-90. 9) Yamada M, Koshika M, Goto S, et al. Lung cancer which accompanies anomalous venous connections, pulmonary and systemic-partial anomalous pulmonary venous connection and persistent left superior vena cava: report of 2 cases. Kyobu Geka 2005; 58: 399-402. (in Japanese with English abstract)

Ann Thorac Cardiovasc Surg Vol. 23, No. 1 (2017)

10) Sakurai H, Kondo H, Sekiguchi A, et al. Left pneumonectomy for lung cancer after correction of contralateral partial anomalous pulmonary venous return. Ann Thorac Surg 2005; 79: 1778-80. 11) Sasaki H, Naka N, Kitahara N, et al. Right partial anomalous pulmonary venous connection found during lobectomy for coexisting lung cancer and tuberculosis: report of a case. Clin Lung Cancer 2006; 7: 350-2. 12) Tanaka R, Nakazato Y, Fukura H, et al. Partial anomalous pulmonary venous connection in right lung cancer: report of a case. Surg Today 2008; 38: 147-9. 13) Chikaishi Y, Nose N, Ichiki Y, et al. A case of partial anomalous pulmonary venous connection in left lung cancer. The journal of the Japanese Association for Chest Surgery 24: 704-9. (in Japanese with English abstract) 14) Hashimoto M, Takuwa T, Okumura Y, et al. A case of Scimitar syndrome with pulmonary sequestration. The Journal of the Japanese Association for Chest Surgery 24: 808-12. (in Japanese with English abstract) 15) Mikubo M, Ikeda S, Hoshino T, et al. Pulmonary resection of lung cancer in a patient with partial anomalous pulmonary venous connection. Ann Thorac Surg 2013; 95: 1799-801. 16) Nagashima T, Tajiri M, Samajima J, et al. Thoracoscopic right upper lobectomy for a pulmonary nodule in a patient with partial anomalous pulmonary venous connection: a case report. Journal of Japan Society for Endoscopic Surgery 18: 363-8. (in Japanese with English abstract) 17) Muranishi Y, Ueshima Y, Muranishi N, et al. A case of partial anomalous pulmonary venous connection in left-lung cancer diagnosed preoperatively. Annals of the Japanease Respiratory Society 2013; 2: 385-8. (in Japanese with English abstract) 18) Asakura K, Izumi Y, Kohno M, et al. Partial anomalous pulmonary venous connection associated with lung cancer in the same lobe: report of a case. Ann Thorac Cardiovasc Surg 2014; 20 Suppl: 457-60. 19) Hashimoto M, Hanaoka J, Oshio Y, et al. Lung squamous cell carcinoma combined with partial anomalous pulmonary venous connect in the same lobe: A case report. The Journal of the Japanese Association for Chest Surgery 2015; 29: 727-31. (in Japanese with English abstract) 20) Tourmousoglou C, Kalogeropoulou C, Koletsis E, et al. Right upper lobe partial anomalous pulmonary venous connection. Case Rep Vasc Med 2014; 2014: 249896.

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Partial Anomalous Pulmonary Venous Connection Coexisting with Lung Cancer: A Case Report and Review of Relevant Cases from the Literature.

A 45-year-old man had an abnormal shadow in the right lung field on an annual screening chest X-ray. He was diagnosed with Stage IA (cT1bN0M0) lung ca...
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