Report an error   


HERDIN Record #: PCHRD12082215451423 Submitted: 22 August 2012 Modified: 24 January 2017

Multiple bilateral pulmonary arteriovenous malformations (PAVMs) rupturing to the left pleura- managed by surgical lobectomy after a failed transcatheter embolization and complicated by postoperative empyema.

Aris Dexter R. Rana,
Francisco M. Peralta Jr.,
Menandro V. Siozon

See More

Pulmonary arteriovenous malformations (PAVM's) are rare pulmonary vascular anomalies. PAVM's consist of abnormal communications between pulmonary arteries and pulmonary veins, causing right-to-left shunting, and thus, frequently with hypoxemia and dyspnea on exertion, although may remain asymptomatic and frequently undiagnosed, unless complications occur. This report presents a PAVM rupturing to the left pleura, a very uncommon clinical problem, but it is an important differential diagnosis of common pulmonary problems such as hemothorax, hypoxemia, and pulmonary nodules. We report a case of a 25 year old female with symptoms of chronic cyanosis and sudden onset of dyspnea. On radiologic evaluations, she was found to have multiple bilateral pulmonary arteriovenous malformations (PAVMs) with multiple blood supply coming from the pulmonary artery. A large PAVM from the left lung that ruptured to the left pleura was documented. An open surgical left lower lobectomy was successfully performed after a failed transcatheter embolization. This was complicated by postoperative empyma a month after hospital discharge which was managed with antibiotics, thoracostomy, open thoracotomy and drainage of empyma and decortication. Patient remained asymptomatic six months after the surgery. Diagnostics and treatement options for multiple PAVMs and management of postoperative empyma are discussed in this paper.

Publication Type
Journal
Publication Sub Type
Case report
Title
The Philippine Journal of Thoracic and Cardiovascular Surgery
Frequency
Annual
Publication Date
December 2007
Volume
14
Page(s)
24-32
LocationLocation CodeAvailable FormatAvailability
Philippine Council for Health Research and Development Library Box No. 8 Fulltext (Request Document)
1. Cottin, V , Dupuis-Girod, S , Lesca, G , Cordier, JF . "Pulmonary arteriovenous malformations in hereditary hemorrhagic telagiectasia: a series of 126 patients" Medicine 86(1): 1-17, 2007. Retrieved from: http://www.ncbi.nlm.nih.gov/pubmed/17220751
2. Cottin, V , Dupuis-Girod, S , et al., . "Pulmonary vascular manifestations of hereditary hemorrhagic telengiectasia (Rendu- Osler Disease)" Respiration 74(4): 361-378, 2007. Retrieved from: http://www.ncbi.nlm.nih.gov/pubmed/17641482
3. Thung, KH , Sihoe, ADL , Wan, IY P, Lee, TW , Wong, R , Yim, AP C. "Hemoptysis from an unusual pulmonary arteriovenous malformation" Ann Thorac Surg 76, 1730-1733, 2003. Retrieved from: http://www.ncbi.nlm.nih.gov/pubmed/14602328
4. Ference, BA , Shannon, TM , White Jr, RI , Zawin, M , Burdge, CM . "Life-theatening pulmonary hemorrhage with pulmonary arteriovenous malformations and hereditary hemorrhagic telangiectasia" Chest J 106, 1387-1390, 1994. Retrieved from: http://www.chestjournal.org/misc/reprints.shtml
5. Laroche, CM , Wells, F , Shneerson, J . "Arteriovenous fistula in pregnancy: massive hemothorax due to enlarging arteriovenous fistula in pregnancy" Chest 101, 1452-1454, 1992. Retrieved from: http://www.chestjournal.org/misc/reprints.shtml
6. Joseph, G , Pati, PK . "Pulmonary arteriovenous malformation: transcatheter embolisation of a large unilateral pulmonary arteriovenous malformation" Heart 89, 731-737, 2003. Retrieved from: http://www.bmjjournals.com/subscriptions/
7. Prasad, V , Chan, RP , Faughnan, ME . "Embolotherapy of pulmonary arteriovenous malformations: efficacy of platinum versus stainless steel coils" J Vasc Interv Radiol 15, 153-160, 2004. Retrieved from: http://www.icvts.org
8. Kohno, M , Noda, K , Moriyama, H , Nishimura, Y . "Segmental arterial ligation undervideo-assisted thoracoscopy combined with transcatheter embolotheraphy for multiple bilateral pulmonary arteriovenous malformations" Int Cardiovasc Thorac Surg , 616-619, 2003. Retrieved from: http://icvts.ctsnetjournals.org/cgi.content/full/3/4/596
9. Remy-Jardin, M , Dumont, P , Brillet, PY , Dupuis, P , Duhamel, A , Remy , J . "Malforamtions treated with embolotherapy: helical CT evaluation of long-term effectiveness after 2-21 year follow-up" J Radiol 239, 576-585, 2006. Retrieved from: http://www.ncbi.nlm.nih.gov/pubmed/16484354
10. Farra, H , Balzer, DT . "Transcatheter occlusion of a large pulmonary arteriovenous malformation using the amplatzer vascular plug" Pediatr Cardiol 26, 683-685, 2005. Retrieved from: http://www.ncbi.nlm.nih.gov/pubmed/16096872
11. Choong, CK , Goodenberg, DM , Picus, D , Meyers, BF . "Surgical treatment of recurrent transient ischemic attacks and hemotysis in a young man with multiple pulmonary arteriovenous malformations" J Thorac Cardiovasc Surg 130, 1456-1458, 2005. Retrieved from: http://jtcs.ctsnetjournals.org/cgi/content/full/130/5/1456
12. Georghiou, GP , Berman, M , Vidne, BA , Saute, M . "Pulmonary arteriovenous malformation treated by lobectomy" Eur J Cardiothorac Surg , 328-330, 2003. Retrieved from: http://elsevier.com/locate/ejcts
13. Bauchner, H . "Empyma thoracis: not time to put down the knife" Arch Dis Child 88, 839, 2003
14. Carey, JA . "Empyma thoracis: a role for open thoracotomy and decortication" Arch Dis Child 79, 510-513, 1998. Retrieved from: http://www.ncbi.nlm.nih.gov/pmc/articles/PMC1717767/
15. Banga, A , Khilnani, GC , Sharma, SK , Dey, AB , Wig, N , Banga, N . "A study of empyma thoracis and role of intrapleural streptokinase in its management" BMC Infect Dis 4, 19, 2004. Retrieved from: http://www.biomedcentral.com/1471-2334/4/19/