Brain Abscess as the First Clinical Manifestation of Isolated Pulmonary Arteriovenous Malformation without Hereditary Hemorrhagic Telangiectasia: A Case Report

腦膿瘍作為非遺傳之出血性血管擴張症的單一肺動靜脈畸形之第一個臨床表徵：個案報告

10.3966/168232812018032801005

鄭暐霖(Wei-Leng Chin)；劉依亭(I-Ting Liu)；林季緯(Chi-Wei Lin)；王浩洸(Hao-Kuang Wang)；孫灼均(Cheuk-Kwan Sun)；洪暐傑(Wei-Chieh Hung)

brain abscess ； pulmonary arteriovenous malformations ； 腦膿瘍 ； 肺動靜脈畸形

台灣家庭醫學雜誌

28卷1期（2018 / 03 / 01）

38 - 44

英文

Pulmonary arteriovenous malformations (PAVMs) are abnormal communications between pulmonary arteries and veins, which may lead to severe complications such as right-to-left shunt, paradoxical systemic embolism, massive pulmonary hemorrhage, cerebrovascular accidents, polycythemia, congestive cardiac failure, and pulmonary hypertension. About 5 to 6% of patients with PAVMs may develop a brain abscess. Most patients with PAVMs also have associated hereditary hemorrhagic telangiectasia (HHT). We present a case of brain abscess secondary to PAVMs, without HHT. A 52-year-old female presented with dizziness and unsteady gait, and was diagnosed as having a brain abscess. To determine the cause of the brain abscess, computed tomography (CT) of the chest with contrast media was performed, which revealed PAVMs with a size of 1.5 cm×1.2 cm in the right middle lobe of the lung. Thus, thoracoscopic wedge resection was performed and pathological findings confirmed the diagnosis. Thus, PAVMs should be considered in patients with brain abscess of unknown etiology. Since PAVMs may not present with pulmonary signs or symptoms, a comprehensive examination is warranted to rule out the disease. Patients with PAVMs should be treated as early as possible to prevent severe complications.

肺動靜脈畸形是一種血管性肺部疾病，為肺動脈和肺靜脈之間的連結異常，可能會導致許多嚴重的併發症，如：右向左分流、反常系統性栓塞、大量肺出血、腦中風、紅血球增多症、鬱血性心衰竭及肺高壓，其中約有5-6%患者可能併發腦膿瘍。多數肺動靜脈畸形的患者會合併遺傳性出血性之血管擴張症，本文個案為單一肺動靜脈畸形。52歲女性，因頭暈及步態不穩而被診斷為腦膿瘍，經過胸部電腦斷層掃描後，發現在右中肺處有一1.5 cm×1.2 cm肺動靜脈畸形，因考慮肺動靜脈畸形可能導致肺膿瘍，因而安排個案接受胸腔鏡肺楔狀切除手術，此個案的重要性在於提供了一個訊息，肺動靜脈畸形的臨床症狀不明顯，因此對腦膿瘍原因不明的病患，亦需考慮進一步詳細檢查，排除是否合併肺動脈畸形所致；若確認為單一肺動脈畸形時的患者，應儘早手術治療以避免致命的併發症。

1. Huang, NC,Tu, CC,Chang, MH,Chen, ST(2008).Pulmonary arteriovenous malformations: a case report.Taiwan J Fam Med,18,48-58.
   連結：
2. Adam, HP,Subbiah, B,Bosch, EP(1977).Neurologic aspects of hereditary hemorrhagic telangiectasia.Arch Neurol,37,101-4.
3. Andersen, PE,Kjeldsen, AD(2010).Interventional treatment of pulmonary arteriovenous malformations.World J Radiol,2,339-44.
4. Anticoli, S,Pezzella, FR,Siniscalchi, A,Gallelli, L,Bravi, MC(2015).Pulmonary arteriovenous malformation as a cause of embolic stroke: case report and review of the literature.Interv Neurology,3,27-30.
5. Babaker, M,Breault, S,Beigelman, C,Lazor, R,Aebischer, N,Qanadli, SD(2015).Endovascular treatment of pulmonary arteriovenous malformations in hereditary haemorrhagic telangiectasia.Swiss Med Wkly,145,w14151.
6. Biçakçioğlu, P,Gülhan, SŞ,Sayilir, E(2017).Surgical treatment of pulmonary arteriovenous malformations.Turk J Med Sci,47,161-6.
7. Corvino, F,Silvestre, M,Cervo, A(2016).Endovascular occlusion of pulmonary arteriovenous malformations with the ArtVentive Endoluminal Occlusion SystemTM.Diagn Interv Radiol,22,463-5.
8. Drøhse, Kjeldsen, A,Tørring, PM,Nissen, H,Andersen, PE(2014).Cerebral abscesses among Danish patients with hereditary haemorrhagic telangiectasia.Acta Neurol Scand,129,192-7.
9. Hsu, CC,Kwan, GN,Thompson, SA,van, Driel, ML(2010).Embolisation therapy for pulmonary arteriovenous malformations.Cochrane Database Syst Rev,5,CD008017.
10. Hwang, CS,Chan, JL(1991).Multiple brain abscesses as a complication of pulmonary arteriovenous fistula. A case report.Changgeng Yi Xue Za Zhi,14,208-12.
11. Kim, HJ,Lee, JS,Oh, YM(2015).Clinical characteristics of pulmonary arteriovenous malformations in Koreans.Respirology,20,155-9.
12. Longo, DL(ed.),Fauci, AS(ed.),Kasper, DL(ed.)(2012).Harrison's Principles of Internal Medicine.New York:McGraw-Hill.
13. Moradi, M,Adeli, M(2014).Brain abscess as the first manifestation of pulmonary arteriovenous malformation: a case report.Adv Biomed Res,3,28.
14. Nakayama, M,Nawa, T,Chonan, T(2012).Prevalence of pulmonary arteriovenous malformations as estimated by low-dose thoracic CT screening.Intern Med,51,1677-81.
15. Nam, TK,Park, Y,Kwon, J(2017).Brain abscesses associated with asymptomatic pulmonary arteriovenous fistulas.J Korean Neurosurg Soc,60,118-24.
16. Shovlin, CL(2014).Pulmonary arteriovenous malformations.Am J Respir Crit Care Med,190,1217-28.
17. Tabakow, P,Jarmundowicz, W,Czapiga, B,Czapiga, E(2005).Brain abscess as the first clinical manifestation of multiple pulmonary arteriovenous malformations in a patient with hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber disease).Folia Neuropathol,43,41-4.
18. Wang, HC,Yang, PC,Kuo, SH,Luh, KT(1998).Pulmonary arteriovenous malformation: analysis of 10 cases.J Formos Med Assoc,97,97-100.