Accepted Manuscript Usefulness of embolization for iatrogenic dural arteriovenous fistula associated with recurrent chronic subdural hematoma: A case report and literature review Tushit Mewada, Tomotaka Ohshima, Taiki Yamamoto, Shunsaku Goto, Yoko Kato PII: S1878-8750(16)30316-3 DOI: 10.1016/j.wneu.2016.05.042 Reference: WNEU 4103 To appear in: World Neurosurgery Received Date: 28 March 2016 Revised Date: 17 May 2016 Accepted Date: 18 May 2016 Please cite this article as: Mewada T, Ohshima T, Yamamoto T, Goto S, Kato Y, Usefulness of embolization for iatrogenic dural arteriovenous fistula associated with recurrent chronic subdural hematoma: A case report and literature review, World Neurosurgery (2016), doi: 10.1016/ j.wneu.2016.05.042. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. Mewada ACCEPTED MANUSCRIPT 1 Usefulness of embolization for iatrogenic dural arteriovenous fistula associated with recurrent RI PT chronic subdural hematoma: A case report and literature review Tushit Mewada1,3, Tomotaka Ohshima2, Taiki Yamamoto2, Shunsaku Goto2, Yoko Kato3 SC 1. Department of Neurosurgery, G B Pant institute of postgraduate medical education and research, M AN U New Delhi, India 2. Department of Neurosurgery, Kariya Toyota General Hospital, Kariya, Japan 3. Department of Neurosurgery, Fujita Health University, Banbuntane Hotokukai Hospital, Nagoya, Japan TE D Corresponding author: Tomotaka Ohshima, MD, 5-15 Sumiyoshi-cho, Kariya, Aichi, 448 8505, EP Japan. Tel: +81-566-21-2450; Fax: +81-566-22-2493; Email: tomotaka.oshima@toyota-kai.or.jp Keywords: Dural arteriovenous fistula, embolization, subdural hematoma, n-butyl cyanoacrylate AC C (NBCA) Abbreviations: dAVF, Dural arteriovenous fistula; DSA, digital subtraction angiography; NBCA, n-butyl cyanoacrylate; MMA, middle meningeal artery; SDH, subdural hematoma Conflicts of interest: None. Mewada ACCEPTED MANUSCRIPT 2 ABSTRACT BACKGROUND: Refractory Chronic subdural hematomas due to iatrogenic dural AVF are difficult RI PT to treat. We report our experience and propose a guideline on basis of literature review for usefulness of embolization of middle meningeal artery (MMA) for the treatment of the same. SC CASE DESCRIPTION: We report the case with right hemiparesis and aphasia 1 month after fall M AN U from bicycle. CT scan head showed left CSDH which was evacuated by burr hole and drainage. The post operative course was complicated by re-accumulation within short period of time. On superselective DSA of MMA iatrogenic dAVF found on left side. We successfully embolized it using NBCA following third irrigation. No re-accumulation found in post operative period till last follow TE D up. We propose treatment protocol based on our experience and literature review. CONCLUSION: Refractory CSDH with re-accumulation with in short interval should be subjected EP to DSA of MMA. Embolization of ipsilateral MMA is safe, effective and useful option for the AC C iatrogenic dAVF treatment and for resolution of hematoma. Mewada ACCEPTED MANUSCRIPT 3 INTRODUCTION Chronic subdural hematoma (CSDH) is relatively common condition in neurosurgery, especially RI PT in old age group on anticoagulants. Burr hole drainage and evacuation of hematoma is perhaps widely accepted management for symptomatic CSDH. Recurrence of CSDH has many etiologies. SC Iatrogenic Dural arteriovenous fistula (dAVF) is rare cause of refractory CSDH. We report our M AN U experience and propose a guideline on basis of literature review for usefulness of embolization of middle meningeal artery (MMA) for the treatment of the same. CASE REPORT TE D A 82 years old female patient presented with right hemiparesis and dysarthria. She had history of minor head trauma in form of fall from bicycle before one month. She was already diagnosed case EP of Alzheimer’s diseases on medications, apart from that no significant medical history, especially AC C anti thrombotic drugs. On evaluation with head CT left side CSDH diagnosed (figure 1a). The CSDH measured approximately 2 cm in maximum thickness. The patient underwent burr-hole drainage of the SDH. On 1st post operative day head CT scan revealed improvement(figure 1b). The recovery was uneventful until 3 weeks postoperatively when she noted reappearance of symptoms. A head CT scan demonstrated recurrence of SDH(figure 2a). The patient was re-operated with burr hole drainage of hematoma with subdural drain placement for 48 hours. A 1st postoperative day head Mewada ACCEPTED MANUSCRIPT 4 CT scan showed improvement (figure 2b). The patient did well postoperatively for 1 month, then again deterioration of symptoms noted. A head CT scan revealed re-accumulation of the SDH(figure RI PT 2c). On evaluating clotting parameters like plasma thromboplastin, partial thromboplastin time, platelet count, factor VIII, fibrinogen, and fibrin-split products, were all normal. Digital subtraction SC angiography (DSA) was performed to rule out an occult vascular malformation leading to repeated M AN U accumulations (figure 3a). Selective left middle meningeal artery injections revealed dAVF just inferior to the burr hole with cotton wool like staining surrounding the burr hole. Because of further recurrence within the month, a third burr hole irrigation with subdural drain placement was Embolization procedure TE D performed. Subsequently, embolization of the MMA was scheduled. A 6F guiding catheter was inserted from the right femoral artery and placed in the left external EP carotid artery. A microcatheter (Marathon, Medtronic, Minneapolis, USA) with micro guidewire AC C (Chikai 10, Asahi intecc, Aichi, Japan) was advanced into the parietal branch of the left MMA. Subsequent angiography confirmed DSA findings of cotton wool-like staining surrounding burr hole with parietal branch of left MMA as feeder and draining veins at inferior end of the burr hole (figure 3b). Fourteen per cent NBCA (Histoacryl; B. Braun, Melsungen, Germany) was prepared with pure NBCA and lipiodol (Lipiodol Ultra-Fluide®, Guerbet, Roissy, France) in 1:6 proportion, was injected after confirmation of the negative result of a provocative test with 1% lidocaine injection. Mewada ACCEPTED MANUSCRIPT 5 The frontal branch was also embolized with 14% NBCA in a similar procedure (figure 3c). Post procedure course RI PT The hematoma started to decrease in size 1 week after embolization and disappeared in 3 months SC (figure 4). No recurrence has been observed since then. M AN U DISCUSSION Chronic subdural hematoma (CSDH) is relatively common condition in neurosurgery, especially in old age group on anticoagulants. The treatment comprises mainly 1 or 2 burr hole irrigation, usually with satisfactory outcome. However, in elderly patients, patients with cerebral atrophy, blood TE D coagulation disorders, and patients on oral antiplatelet/anticoagulant drugs, resolution may not be achieved even after several rounds of surgery, and CSDH is sometimes difficult to treat. 1 There are EP many etiologies for the recurrence like chronic alcoholism, cerebral atrophy, hepatic dysfunction, AC C use of oral anticoagulant drugs, hemodialysis, blood coagulation disorder, shunt surgery, post operative infection and rarely dAVF. 1,2,3 Trivial trauma is most common cause of CSDH in elderly patients, leading to rupture of bridging vein and accumulation of blood products mixed CSF between two layers of dura. Recurrent bleed in outer membrane capillaries thought to be cause of enlargement of CSDH. Histologically the outer membrane and dura mater have capillary like communicating vessels, which penetrate through the Mewada ACCEPTED MANUSCRIPT 6 dura mater and connect to MMA.4 These can be seen as cotton wool like staining or visualization of scattered abnormal vascular network using Superselective DSA of MMA. In refractory recurrent RI PT cases DSA shows diffuse dilatation of MMA near CSDH membrane and burr hole.5,6 Overall these evidences support role of embolization of the MMA for interrupting the blood supply to the outer SC membrane and stop recurrence of hematoma. M AN U Iatrogenic dAVF are rarity. 7 Anatomically two meningeal veins normally accompany the MMA and its major branches. Interestingly, the veins rather then arteries, occupy the meningeal groove in skull bone.7,8 Injury at this region leads to abnormal communication and eventually dAVF, producing characteristic tram tracks appearance on DSA. The same phenomenon can be demonstrated in our TE D case as seen in figure 3b. It shows iatrogenic dAVF after burr hole placement treated successfully with embolization of ipsilateral MMA. EP Embolization of the MMA is highly useful for refractory CSDH as per literature review.1,5,6,9,10 AC C Tempaku et al10 showed efficacy of embolization of MMA in five patients. Hashimoto et al 1 and Mino et al 9 also support use of embolization of MMA in refractory CSDH. The commonly used embolic materials are NBCA, polyvinyl alcohol particles, and coils with almost same therapeutic outcomes.1,6,10 We used NBCA due to its low recanalization rate and liquid state providing suitability for the peripheral use. Mewada ACCEPTED MANUSCRIPT 7 Subdural drain placement is very effective method in prevention of recurrence.11 The dAVF associated recurrence is usually refractory to this as outer membrane capillaries receive high flow RI PT inputs from dAVF. Intra-operative coagulation of the membrane might be insufficient sometimes. Prevention of iatrogenic dAVF can be done by using fusion images in preoperative planning but SC efficacy of the method is questionable due to reconstruction artifact and anatomical variations.5,6 M AN U We propose a protocol based on literature review that patients with refractory CSDH, i.e. two or more recurrences after satisfactory treatment and re accumulation with in short interval i.e. within 1 month, should be subjected to DSA of external carotid arteries. If dAVF found, embolization of TE D ipsilateral MMA is safe, effective and beneficial treatment option. EP CONCLUSION AC C Refractory CSDH with re accumulation with in short interval should be subjected to DSA of MMA. Embolization of ipsilateral MMA is safe, effective and useful option for the iatrogenic dAVF treatment and for resolution of hematoma. Mewada ACCEPTED MANUSCRIPT 8 REFERENCES 1. Hashimoto T, Ohashi T, Watanabe D, Koyama S, Namatame H, Izawa H, et al. Usefulness RI PT of embolization of the middle meningeal artery for refractory chronic subdural hematomas. Surg Neurol Int. 2013;4:104-07. SC 2. Chon KH, Lee JM, Koh EJ, Choi HY. Independent predictors for recurrence of chronic M AN U subdural hematoma. Acta Neurochir (Wien). 2012;154:1541-8. 3. Leroy HA, Aboukaïs R, Reyns N, Bourgeois P, Labreuche J, Lejeune JP. Predictors of functional outcomes and recurrence of chronic subdural hematomas. J Clin Neurosci. 2015;22:1895-900. TE D 4. Tanaka T, Kaimori M. Histological study of vascular structure between the dura mater and the outer membrane in chronic subdural hematoma in an adult. No Shinkei Geka. EP 1999;27:431‐6. AC C 5. Takahashi K, Muraoka K, Sugiura T, Maeda Y, Mandai S, Gohda Y, et al. Middle meningeal artery embolization for refractory chronic subdural hematoma: 3 case reports. No Shinkei Geka. 2002;30:535‐9. 6. Tanaka T, Fujimoto S, Saitoh K, Satoh S, Nagamatsu K, Midorikawa H. Superselective angiographic findings of ipsilateral middle meningeal artery of chronic subdural hematoma in adults. No Shinkei Geka. 1998;26:339-47. Mewada ACCEPTED MANUSCRIPT 9 7. Pappas CT, Zabramski JM, Shetter AG. Iatrogenic arteriovenous fistula presenting as a recurrent subdural hematoma. Case report. J Neurosurg. 1992;76:134-6. RI PT 8. Feldman RA, Hieshima G, Giannotta SL. Traumatic dural arteriovenous fistula supplied by scalp, meningeal, and cortical arteries: case report. Neurosurgery 1980;6: 670-74. SC 9. Mino M, Nishimura S, Hori E, Kohama M, Yonezawa S, Midorikawa H, et al. Efficacy of M AN U middle meningeal artery embolization in the treatment of refractory chronic subdural hematoma. Surg Neurol Int. 2010;1:78-81. 10. Tempaku A, Yamauchi S, Ikeda H, Tsubota N, Furukawa H, Maeda D, et al. Usefulness of interventional embolization of the middle meningeal artery for recurrent chronic subdural TE D hematoma: Five cases and a review of the literature. Interv Neuroradiol. 2015;21:366-71. 11. Santarius T, Kirkpatrick PJ, Ganesan D, Chia HL, Jalloh I, Smielewski P, et al. Use of EP drains versus no drains after burr-hole evacuation of chronic subdural haematoma: a AC C randomised controlled trial. Lancet. 2009; 374: 1067–73. Mewada 10 RI PT ACCEPTED MANUSCRIPT SC Figure Legends day. M AN U Fig. 1. Non contrast head CT on admission (a) showing left CSDH and (b) on first post operative Fig. 2. (a) CT scan after 1 month of first irrigation and (b) after second evacuation. (c) CT scan showing re-recurrence after second irrigation. TE D Fig. 3. (a) Superselective DSA of left MMA with area of interest in red rectangle, (b) enlarging the area of interest showing cotton wool area surrounding burr hole, feeders from left MMA and point of EP fistula at lower border of burr hole with corresponding draining veins. (c) showing corresponding AC C DSA after embolization of left MMA, disappearance of the dAVF. Fig. 4. Non contrast head CT scan after 3 months of left MMA embolization with burr hole drainage AC C EP TE D M AN U SC RI PT ACCEPTED MANUSCRIPT figure 1: a and b AC C EP TE D M AN U SC RI PT ACCEPTED MANUSCRIPT M AN U SC RI PT ACCEPTED MANUSCRIPT AC C EP TE D figure 2 : a, b, c, AC C EP TE D M AN U SC RI PT ACCEPTED MANUSCRIPT M AN U SC RI PT ACCEPTED MANUSCRIPT AC C EP TE D figure 3: a ,b , c EP AC C figure 4 TE D M AN U SC RI PT ACCEPTED MANUSCRIPT ACCEPTED MANUSCRIPT HIGHLIGHT Refractory CSDH due to iatrogenic dural AVF are difficult to treat. • Refractory CSDH with re-accumulation with in short interval should be subjected to DSA RI PT • of MMA. SC Embolization of ipsilateral MMA is safe, effective and useful option for the iatrogenic EP TE D M AN U dAVF treatment. AC C •