Interventional embolization therapy for complex pulmonary arteriovenous fistula: case report and literature review_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

SHI Fangyu , JIANG Faming , WANG Lan , WANG Ye ,  LIANG Zongan

Department of Respiratory and Critical Care Medicine, West China Hospital, Sichuan University, Chengdu, Sichuan 610041, P. R. China;

Corresponding author：

LIANG Zongan, Email: liang.zongan@163.com

Keywords：

Complex pulmonary arteriovenous fistula; Clinical features; Interventional embolization therapy

DOI：

10.7507/1671-6205.201804064

Video：

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Objective To explore the clinical characteristics and improve the knowledge of diagnosis and treatment of complex pulmonary arteriovenous fistula (PAVF) as well as enrich the experience of diagnosis and treatment of the disease.Methods A retrospective analysis of pathogenetic process clinical manifestations, imaging features and diagnosis and treatment was conducted on one case of complex PAVF. The literature review was carried out with " complex pulmonary arteryovenous fistula (malformation)” as the research terms in English and Chinese respectively in CNKI, WanFang and PubMed database. Search time ranged from January 1997 to April 2018, and the literature was screened and reviewed.Results The patient was a 47-year-old female complained of recurrent epistaxis for 40 years, intermittent hemoptysis for 20 years, headache, dizziness, chest pain, chest tightness for 4 years and the symptoms were aggravated by 3 months, visiting this hospital on January 23, 2018. Pulmonary CT angiography revealed multiple nodules in internal and external segment and outer basal segment of right lung, anterior basal segment and outer basal segment of left lung. CT enhanced scan showed that the thickened pulmonary artery was connected with the above lesion, and the edge was accompanied by large draining veins. Pulmonary artery revascularization showed complex PAVF abnormal branches. The diagnosis was complex PAVF, and interventional embolization therapy was carried out and curative effect was satisfactory during the follow-up. A total of 6 literatures were reviewed in above-mentioned databases, including 4 Chinese literatures and 2 English literatures, containing 10 patients, including 8 males and 2 females, with an average age of (9.7±7.0) years. Most of the clinical manifestations were shortness of breath after exercise, cyanosis and hemoptysis and all patients were cured and discharged after interventional embolization treatment except for 1 patient refused treatment.Conclusions Complex PAVF is a very rare pulmonary vascular malformation. The clinical manifestations mainly include hypoxemia, dyspnea, hemoptysis, and the preferred treatment is interventional embolization, which has a satisfactory clinical effect at a short-term follow-up.

Citation： SHI Fangyu, JIANG Faming, WANG Lan, WANG Ye, LIANG Zongan. Interventional embolization therapy for complex pulmonary arteriovenous fistula: case report and literature review. Chinese Journal of Respiratory and Critical Care Medicine, 2019, 18(2): 156-161. doi: 10.7507/1671-6205.201804064 Copy

1.	Lacombe P, Lacout A, Marcy PY, et al. Diagnosis and treatment of pulmonary arteriovenous malformations in hereditary hemorrhagic telangiectasia: an overview. Diagn Interv Imaging, 2013, 94(9): 835-848.
2.	Aggarwal V, Khan DM, Rhodes JF. Pulmonary arteriovenous malformation causing systemic hypoxemia in early infancy. Case Rep Pediatr, 2017, 2017: 2841720.
3.	Hundt W, Kalinowski M, Kiessling A, et al. Novel approach to complex pulmonary arteriovenous malformation embolization using detachable coils and Amplatzer vascular plugs. Eur J Radiol, 2012, 81(5): e732-e738.
4.	Ahn S, Han J, Kim HK, et al. Pulmonary arteriovenous fistula: clinical and histologic spectrum of four cases. J Pathol Transl Med, 2016, 50(5): 390-393.
5.	杨舟, 陈智, 向金星, 等. 介入堵塞治疗儿童复杂型肺动静脉瘘. 临床儿科杂志, 2014, 32(10): 964-964.
6.	Pradhan A, Khare R, Sethi R. A rare case of congenital complex pulmonary AV fistula. BMJ Case Rep, 2014, 10: pii: bcr2014205939.
7.	Lacombe P, Lagrange C, Hajjam ME, et al. Reperfusion of complex pulmonary arteriovenous malformations after embolization: report of three cases. Cardiovasc Intervent Radiol, 2005, 28(1): 30-35.
8.	杨呈伟, 李炯佾, 吴文辉, 等. 介入栓塞治疗囊状复杂型肺动静脉瘘一例. 放射学实践, 2010, 25(10): 1177-1178.
9.	蒲晓群, 黄万喜, 邓金华, 等. 经导管植入弹簧钢圈栓塞治疗复杂型肺动静脉瘘 1 例. 临床心血管病杂志, 1999, 15(12): 574.
10.	王执民, 耿斌, 左健, 等. 用弹簧栓子栓塞治疗幼儿复杂型肺动静脉瘘 (附 1 例报告). 实用放射学杂志, 1996, 12(11): 673-674.
11.	Moussouttas M, Fayad P, Rosenblatt M, et al. Pulmonary arteriovenous malformations cerebral ischemia and neurologic manifestations. Neurology, 2000, 55(7): 959-964.
12.	Abdel Aal AK, Eason J, Moawad S, et al. Persistent pulmonary arteriovenous malformations: percutaneous embolotherapy. Curr Probl Diagn Radiol, 2017, 47(6): 428-436.
13.	Pollak JS, Saluja S, Thabet A, et al. Clinical and anatomic outcomes after embolotherapy of pulmonary arteriovenous malformations. J Vasc Interv Radiol, 2006, 17(1): 35-44.
14.	Nakamura H, Miwa K, Haruki T, et al. Pulmonary arteriovenous fistula with cerebral infarction successfully treated by video-assisted thoracic surgery. Ann Thorac Cardiovasc Surg, 2008, 14(1): 35-37.
15.	Gallitelli M, Lepore V, Pasculli G, et al. Brain abscess: a need to screen for pulmonary arteriovenous malformations. Neuroepidemiology, 2005, 24(1-2): 76-78.
16.	Tørring PM, Kjeldsen AD, Ousager LB, et al. ENG mutational mosaicism in a family with hereditary hemorrhagic telangiectasia. Mol Genet Genomic Med, 2018, 6(1): 121-125.
17.	Shovlin CL, Guttmacher AE, Buscarini E, et al. Diagnostic criteria for hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber syndrome). Am J Med Genet, 2000, 91(1): 66-67.
18.	Richards-Yutz J, Grant K, Chao EC, et al. Update on molecular diagnosis of hereditary hemorrhagic telangiectasia. Hum Genet, 2010, 128(1): 61-77.
19.	Natarajan K, Khabiri H, Jung S. Case 3. Pulmonary arteriovenous malformation (PAVM): paradoxical embolism through the arteriovenous fistula can cause brain abscess and infarct. Am J Roentgenol, 2000, 175(3): 854, 857-858.
20.	Winterkorn JM, Kupersmith MJ, Wirtschafter JD, et al. Brief report: treatment of vasospastic amaurosis fugax with calcium-channel blockers. N Engl J Med, 1993, 329(6): 396-398.

1. Lacombe P, Lacout A, Marcy PY, et al. Diagnosis and treatment of pulmonary arteriovenous malformations in hereditary hemorrhagic telangiectasia: an overview. Diagn Interv Imaging, 2013, 94(9): 835-848.
2. Aggarwal V, Khan DM, Rhodes JF. Pulmonary arteriovenous malformation causing systemic hypoxemia in early infancy. Case Rep Pediatr, 2017, 2017: 2841720.
3. Hundt W, Kalinowski M, Kiessling A, et al. Novel approach to complex pulmonary arteriovenous malformation embolization using detachable coils and Amplatzer vascular plugs. Eur J Radiol, 2012, 81(5): e732-e738.
4. Ahn S, Han J, Kim HK, et al. Pulmonary arteriovenous fistula: clinical and histologic spectrum of four cases. J Pathol Transl Med, 2016, 50(5): 390-393.
5. 杨舟, 陈智, 向金星, 等. 介入堵塞治疗儿童复杂型肺动静脉瘘. 临床儿科杂志, 2014, 32(10): 964-964.
6. Pradhan A, Khare R, Sethi R. A rare case of congenital complex pulmonary AV fistula. BMJ Case Rep, 2014, 10: pii: bcr2014205939.
7. Lacombe P, Lagrange C, Hajjam ME, et al. Reperfusion of complex pulmonary arteriovenous malformations after embolization: report of three cases. Cardiovasc Intervent Radiol, 2005, 28(1): 30-35.
8. 杨呈伟, 李炯佾, 吴文辉, 等. 介入栓塞治疗囊状复杂型肺动静脉瘘一例. 放射学实践, 2010, 25(10): 1177-1178.
9. 蒲晓群, 黄万喜, 邓金华, 等. 经导管植入弹簧钢圈栓塞治疗复杂型肺动静脉瘘 1 例. 临床心血管病杂志, 1999, 15(12): 574.
10. 王执民, 耿斌, 左健, 等. 用弹簧栓子栓塞治疗幼儿复杂型肺动静脉瘘 (附 1 例报告). 实用放射学杂志, 1996, 12(11): 673-674.
11. Moussouttas M, Fayad P, Rosenblatt M, et al. Pulmonary arteriovenous malformations cerebral ischemia and neurologic manifestations. Neurology, 2000, 55(7): 959-964.
12. Abdel Aal AK, Eason J, Moawad S, et al. Persistent pulmonary arteriovenous malformations: percutaneous embolotherapy. Curr Probl Diagn Radiol, 2017, 47(6): 428-436.
13. Pollak JS, Saluja S, Thabet A, et al. Clinical and anatomic outcomes after embolotherapy of pulmonary arteriovenous malformations. J Vasc Interv Radiol, 2006, 17(1): 35-44.
14. Nakamura H, Miwa K, Haruki T, et al. Pulmonary arteriovenous fistula with cerebral infarction successfully treated by video-assisted thoracic surgery. Ann Thorac Cardiovasc Surg, 2008, 14(1): 35-37.
15. Gallitelli M, Lepore V, Pasculli G, et al. Brain abscess: a need to screen for pulmonary arteriovenous malformations. Neuroepidemiology, 2005, 24(1-2): 76-78.
16. Tørring PM, Kjeldsen AD, Ousager LB, et al. ENG mutational mosaicism in a family with hereditary hemorrhagic telangiectasia. Mol Genet Genomic Med, 2018, 6(1): 121-125.
17. Shovlin CL, Guttmacher AE, Buscarini E, et al. Diagnostic criteria for hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber syndrome). Am J Med Genet, 2000, 91(1): 66-67.
18. Richards-Yutz J, Grant K, Chao EC, et al. Update on molecular diagnosis of hereditary hemorrhagic telangiectasia. Hum Genet, 2010, 128(1): 61-77.
19. Natarajan K, Khabiri H, Jung S. Case 3. Pulmonary arteriovenous malformation (PAVM): paradoxical embolism through the arteriovenous fistula can cause brain abscess and infarct. Am J Roentgenol, 2000, 175(3): 854, 857-858.
20. Winterkorn JM, Kupersmith MJ, Wirtschafter JD, et al. Brief report: treatment of vasospastic amaurosis fugax with calcium-channel blockers. N Engl J Med, 1993, 329(6): 396-398.

Chinese Journal of Respiratory and Critical Care Medicine

Interventional embolization therapy for complex pulmonary arteriovenous fistula: case report and literature review

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