· 既往史：糖尿病控制不佳，高血压，无服用避孕药史。

· 体格检查：GCS 15, 肌肉力量III-IV，肌肉张力增加：齿轮样肌强直。认知障碍（MoCA 1/30，MMSE 8/30）。

· 腰椎穿刺显示脑脊液压力为200mmh₂O。

Figure 1. SWI revealed cortical and deep venous ectasia (blue arrow) and hyperintensive signal of sinus (red arrow).

图 1. SWI显示皮层静脉和深部静脉扩张（蓝箭）以及静脉窦内高信号（红箭）。

Figure 2. T2 Flair showed multiple hyperintensity of bilateral coronal radiation, paraventricular, bilateral occipital lobes and left cerebellum, indicating venous cerebral edema.

图 2. T2-Flair显示双侧放射冠、侧脑室旁、双侧枕叶及左侧小脑多发高信号，提示静脉源性脑水肿。

Figure 3. DWI showed multiple hyperintensity of bilateral coronal radiation, paraventricular, occipital lobes and left cerebellum, indicating cytotoxic cerebral edema.

图 3. DWI显示双侧放射冠、侧脑室旁、双侧枕叶及左侧小脑多发高信号，提示细胞毒性脑水肿。

Figure 4 GIF. The DAVF fed by MMA and occipital artery, drained into the ipsilateral sigmoid sinus and refluxed to the straight sinus, lateral sinus, SSS, and the vein of Labbe (Cognard IIa b).

图 4 GIF. DAVF由MMA和枕动脉供血，引流至同侧侧窦， 对侧侧窦、直窦和SSS以及对侧Labbe静脉（Cognard IIa b）。

Figure 5. 左侧横-乙交界区弥散的动静脉瘘，血管结构尚不清晰。右侧枕动脉发出的硬膜分支在窦汇区移行为硬膜静脉，向左侧横-乙交界区汇集。

图 5. The angioarchitecture of the diffusive fistula located at the left transverse-sigmoid junction was not demonstrated clearly. Meningeal branches arose from the right occipital artery transferred in to a dural venous channel, and converged into the left transverse-sigmoid junction fistula.

Figure 6. Meningeal branches originated from the MCA and SCA also joined the fistula.

图 6. MCA和SCA发出的硬膜支参与硬脑膜动静脉瘘供血。

Figure 7 GIF. Severe stenosis at the left jugular foramen induced drainage deficit and forced the fistula refluxed to upstream sinuses.

图 7 GIF. 左侧乙状窦颈静脉孔段重度狭窄导致静脉回流障碍，迫使瘘向上游静脉窦逆流。

Figure 8. The tentorial and vermian groups of cerebellar venous drainage was obstructed due to sinus hypertension, which was compensated via the right petrous bridging vein and anterior pontomesencephalic venous system.

图 8. 静脉窦高压导致小脑幕和蚓部的静脉回流受阻，通过右侧岩部桥静脉和桥-中脑前静脉系统进行代偿。

Figure 9. Severe stenosis (or near occlusion) of the right transverse-sigmoid junction aggravated the drainage deficit, and induced reflux to the right vein of Labbe.

图 9. 右侧侧窦狭窄/近乎闭塞病变加重了全脑的静脉回流障碍,并向右侧Labbe 静脉逆流。

Figure 10. The functional drainage of the SSS was occupied by the fistulous reflux, compensated via the deep venous system and diploic veins.

图 10. 上矢状窦的功能性回流被瘘的竞争性反流占据，通过深部静脉及板障静脉代偿。

1

Angioarchitecture and strategies

The fistula was fed by MMA, occipital artery and dural branches of the pial arteries, the posterior branch of MMA is the optimal TAE route.

The venous stenosis of the ipsilateral sigmoid sinus at the jugular foramen restricted the antegrade drainage and caused the fistula reflux.

–However, venous stenting at the jugular foramen has higher restenosis rate.

–The right lateral sinus reconstruction can provide more venous drainage capacity rapidly and relieve the straight and SSS reflux.

The clinical symptoms aggravated with deterioration of consciousness after the angiography, is related to severe drainage deficit without efficient compensation. Anticoagulation therapy (heparin) was administered immediately.

Sinus reconstruction is our prior option, which would provide opportunity of TAE by increasing the drainage compensation.

Retrograde recanalization of each sinuses respectively;

Stenting of the left sigmoid sinus can compress the dural venule and prevent Onyx from penetrating into the sinus lumen.

Dual antiplatelet therapy was administered for 5 days before sinus stenting.

DAVF主要由MMA、枕动脉和邻近软膜动脉的硬膜支供血，脑膜中动脉后支是首选的经动脉治疗路径。

患侧乙状窦颈静脉孔段的重度狭窄限制了顺向引流，并引起反流。

–患侧乙状窦颈静脉孔段的重度狭窄限制了顺向引流，并引起反流。

–而右侧侧窦重建可迅速提供更多静脉回流的代偿能力，并改善直窦和上矢状窦的逆流。

血管造影术后患者即出现明显症状加重，表现为意识水平下降，与严重的静脉回流障碍且缺乏有效的静脉侧枝代偿相关，故即刻给予规范的抗凝治疗（肝素）。

静脉窦重建是我们的首选方案，这将增加引流代偿，为实施TAE提供机会。

双侧静脉窦均采用逆行方向直接开通。

左乙状窦支架可以通过压迫硬膜小静脉减少瘘口流量，通过一定程度上防止Onyx直接进入窦腔。

静脉窦支架的术前用药：双抗血小板治疗5天。

2

Treatment process

Figure 11. The origin of the sinus tributaries should be minded before venous access: the petrous group of the cerebellar veins converged to the proximal sigmoid sinus. The reflux to the vein of Labbe was originated from the TS.

图 11. 静脉入路时应注意静脉窦属支的起源:小脑岩静脉组汇聚到乙状窦近端。Labbe静脉回流到TS。

Figure 12. The right transverse-sigmoid stenosis was recanalized first. 6F Long sheath was placed at the superior bulb of JIV. Envoy DA was advanced to the transverse-sigmoid stenosis. A loop guidewire is preferred to prevent punctuation, however.

图 12. 首先处理右侧横-乙交界的狭窄；6F长鞘置于右侧颈静脉上球部，Envoy DA在置于横-乙交界 病变近端。如果导丝不能直接通过病变段，则首选成袢通过，可以防止刺破静脉窦属支。

Figure 13 GIF. Minor subdural hemorrhage due to venule rupture during venography. Withdrew the guiding catheter can not demonstrate distal lumen.  Microcatheter venography revealed dural venule. Envoy DA passed into narrow segment by Echelon-10.

图 13 GIF. 高压注射器静脉造影可见小静脉破裂引起的少量硬膜下出血。回撤引导导管无法显示远端管腔。动静脉联合路途下，Echelon10通过病变段后，同时加用0.035导丝支撑，将DA通过病变段。

Figure 14 GIF. Another working projection to prevent penetrate into the dural venule. Due to previous minor subdural hemorrhage, we chose a more conservative small balloon. Venous sinus angioplasty was performed by LitePac 4*30.

图 14 GIF. 更换工作角度，以防止穿破硬膜小静脉。由于之前少量的硬膜下出血，我们保守地选择较小的LitePac 4*30球囊扩张狭窄段。

Figure 15. Precise 8*40mm was placed at stenotic segment. A larger LitePac 6*20mm balloon was dilated post stenting dilation. The Envoy DA can access through the recanalized segment, venography revealed no reflux to the vein of Labbe and no hemorrhage.

图 15. Precise 8*40mm支架于狭窄段释放，6mm球囊进行支架后扩张。Envoy DA通过病变段，直接静脉造影显示Labbe静脉无逆流，局部无出血。

Figure 16 GIF. After right lateral sinus reconstruction, the aggravated fistula transferred from Cognard 2a b to a benign Cognard 2a fistula. No reflux to the SSS, straight sinus or the right vein of Labbe.

图 16 GIF. 右侧侧窦重建后，丛侵袭性（Cognard 2a b）转变成良性DAVF（Cognard 2a）。上矢状窦、直窦及右侧Labbe静脉逆流消失。

Figure 17. Minor subdural hemorrhage (orange arrows),  the stent was expended well.

图 17. 少量硬膜下出血（橙色箭头），支架扩张良好。

Figure 18. The stenosis at the left jugular foramen was expanded with a 4mm balloon.

图 18. 以4mm球囊行左侧颈静脉孔狭窄段扩张。

Figure 19 GIF. The Envoy DA was advanced to the real lumen of the TS. Sinus stent covered the shunted pouch (orange circle).The proximal side of the stent expanded unsatisfactorily, and can not be fully expanded with a 6mm balloon.

图 19 GIF. Envoy DA置于横窦真腔内。静脉窦支架覆盖左侧的窦前静脉间隔（橙色虚线）。支架近端扩张欠满意，且6mm球囊后扩张亦可见束腰征。

Figure 20 GIF. A 7*40mm Precise stent was deployed tandemly on the proximal side. Residual stenosis was considered caused by a venous septum: the inferior portion (orange circle) is the recipient venous pouch, while the superior portion is the drainage lumen.

图 20 GIF. Precise7*40mm支架在乙状窦近端串联释放。残余狭窄考虑为静脉间隔导致：下方为瘘的受体静脉间隔，而上方为正常的引流间隔。

Figure 21. The relationship between the dural venule, the shunted pouch (orange arrows) and sinus wall is demonstrated clearly on dual volume reconstruction: the dural veins converge to a single or limited venous channels before empty into the sinus.

图 21. 双容积重建可显示硬膜小静脉、窦前间隙（橙色箭头）和窦壁之间的关系：硬膜静脉先汇聚为共同的静脉通道再进入静脉窦内。

Figure 22. TAE with Marathon microcatheter was performed via the posterior branch of the MMA.

图 22. Marathon微导管通过脑膜后动脉到达窦壁行经动脉栓塞。

Figure 23. The Onyx cast in the dural veins instead of penetrating to the sinus lumen.

图 23. Onyx在窦前的硬膜静脉内铸型，而非穿支架网眼进入静脉窦。

Figure 24. The Onyx cast in the petrous veins (blue arrow) , the Onyx should be solidify cast in the shunted pouch (orange arrow).

图 24. Onyx沿岩部硬膜静脉铸型，此时还应在窦前间隙（橙色箭头）更多铸型。

Figure 25. The dural fistulous veins converged to the shunted pouch (orange arrows).

图 25. 该“窦前间隙”（橙色箭头）是硬膜静脉的汇聚点。

Figure 26. The left sigmoid sinus was patient after TAE.

图 26. 静脉窦腔内保持通畅。

Figure 27 GIF. Venous stasis significantly improved post-operation.

图 27 GIF. 术后静脉瘀滞明显改善。

Figure 28 GIF. The residual fistula can be treated via MMA again if necessary.

图 28 GIF. 一期手术的终点，是静脉窦回流障碍改善，尽管残余瘘口仍存在窦逆流，但对脑组织的正常回流影响很小。

3

Follow-Up

Figure 29 GIF. Reflux to contralateral sinus (Cognard IIa).

图 29 GIF. 术后随访DAVF向对侧侧窦引流（Cognard IIa）。

‍Figure 30. No significant increased DAVF flow during follow-up process.

图 30. 随访过程中DAVF血流未见明显增加。

Figure 31 GIF. No residual feeders from pial artery, while cerebral drainage improved significantly.

图 31 GIF. 颅内引流显著改善，未见残余软膜供血动脉。

Figure 32. The feeding posterior meningeal artery disappeared, and posterior venous drainage gradually improved.

图 32. 脑膜后动脉供血支消失，后循环静脉引流改善。

Figure 33. T2 Flair showed hyperintensity decreased gradually, indicating venous cerebral edema relieved.

图 33. T2-Flair显示高信号逐渐消失，提示静脉源性水肿缓解。

Figure 34. DWI showed hyperintensity decreased gradually, indicating cytotoxic edema had been relieved.

图 34. DWI显示高信号逐渐消失，提示细胞毒性水肿得到缓解。

Figure 35 GIF. 15M FU PE: bilateral limb muscle strength V, no limb trembling. Normal cognition, no intracranial pulsatile tinnitus. 
Pre    MoCA 1/30,   MMSE 8/30 
Post       MoCA 6/30, MMSE 6/30 
3M FU     MoCA 17/30, MMSE 22/30 

15M FU    MoCA 16/30, MMSE 23/30

图 35 GIF. 15月随访体格检查：双侧肢体肌力V，无肢体震颤，精细动作恢复良好。认知功能恢复，无颅内杂音。

术前    MoCA 1/30,   MMSE 8/30 

术后       MoCA 6/30, MMSE 6/30 

三个月随访     MoCA 17/30, MMSE 22/30 

十五个月随访    MoCA 16/30, MMSE 23/30

4

Summary

The clinical symptoms aggravated with deterioration of consciousness after the angiography, is related to severe drainage deficit without efficient compensation. The severe stenoses of the contralateral transverse-sigmoid junction and ipsilateral jugular foremen induced fistulous reflux to the straight and SSS and the right vein of Labbe. Therefore sinus reconstruction is prior to fistula embolization, which can transfer the aggressive high-grade DAVF into a low-grade benign fistula.

For sinus reconstruction, the right lateral sinus recanalization can increase the compensation capacity rapidly, and has higher long-term patency rate. Meanwhile, stenting across the jugular foramen has higher re-stenotic rate. Therefore the right sinus reconstruction was performed prior to the left side.

The stenting of the right stenotic sinus resolved the obstructive drainage deficit, the fistula transferred to Conard 2a immediately, which has a more benign natural prognosis. Further embolization is aimed to decrease the fistulous flow and competitive drainage disorder. The curative embolization of the shunt is NOT our target.

The shunted pouch adjacent to the transverse-sigmoid junction is where the fistulous veins converged area, which is the target to obliterate the fistula efficiently. Transvenous embolization, transarterial embolization with sinus protective technique are feasible.

In this case, stents protective technique was performed. The Onyx prone to penetrate at the shunted pouch instead of into the sinus lumen during the TAE.

Direct TVE of the shunted pouch has
a higher obliterate rate of the fistula.

Due to the patient’s movement and cognitive disorder has recovered, no pulsatile tinnitus was identified, and the residual fistula was stable with patent sinus drainage. Therefore, no necessary for further endovascular treatment. Antiplatelet therapy and angiographic follow up was administered.

血管造影术后患者即出现明显症状加重，表现为意识水平下降，与严重的静脉回流障碍且缺乏有效的静脉侧枝代偿相关。对侧横-乙交界和同侧乙状窦颈静脉孔段的严重狭窄导致DAVF向直窦、上矢状窦及侧裂静脉逆流。所以优先开通静脉窦改善静脉回流，使高分级的侵袭性DAVF转变为低分级的良性瘘。

静脉窦重建中，考虑到右侧侧窦开通可以快速改善静脉回流的代偿功能，缓解直窦和上矢状窦的逆流，且支架远期通畅率高。而乙状窦-颈静脉孔段支架置入术后再狭窄率高。故先处理右侧静脉窦病变。

开通右侧侧窦的局部狭窄，解决了梗阻性静脉回流障碍，术前Cognard 2a b的侵袭性DAVF立刻降为Cognard 2a（其自然病程多为良性）。进一步栓塞的目的是降低流量，减少瘘导致的竞争性回流障碍，不必追求完全的解剖学治愈。

左侧横-乙交界区的窦前静脉间隙是瘘的硬膜静脉的主要汇聚点，是高效栓塞瘘口的关键靶点。可以采用颈静脉途径栓塞、经动脉栓塞或辅以静脉窦保护技术。

该病例采用静脉窦支架保护技术，经动脉栓塞时Onyx胶首先在窦前的硬膜静脉汇聚点弥散，而非直接进入静脉窦。

经静脉途径直接进入窦前间隙，可以更高效的闭塞该动静脉瘘。

随访过程中患者临床症状改善，运动、认知功能恢复，无颅内杂音，残余DAVF稳定，静脉回流通畅。故无需进一步治疗干预，建议抗血小板治疗，同时临床及血管造影随访。
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张晓龙

复旦大学附属华山医院

复旦大学附属华山医院放射科主任医师，博士、教授、博士生导师;

斯坦福大学医学院客座临床教授;

主持国家自然科学基金3项，第一作者或通讯作者发表国内外权威期刊文章50余篇;

中华医学会、放射学会、卫生部医政司等组织中担任副主任委员、组长等职务.《中国名医百强榜》神经介入专业中国十强（2012年度、2013年度、2014年度、2015-16年度、2017-18年度）;

擅长复杂和疑难脑血管疾病的介入治疗，如复杂脑动脉瘤的栓塞，硬脑膜动静脉瘘栓塞，脑动静脉畸形栓塞，脑梗死的支架，脊髓血管畸形治疗;

自1995年开始从事脑血管疾病介入诊治工作和研究，师从黄祥龙教授、沈天真教授和凌锋教授，是我国最早从事神经介入的专家之一。2010年9月至今连续介入治疗颅内动脉瘤1500余例,无操作致死.

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