骶管囊肿文献导读：脊髓电刺激治疗骶管囊肿术后慢性盆底疼痛患者病例报告

原文：Spinal cord stimulation for the treatment of chronic pelvic pain after Tarlov cyst surgery in a 66-year-old woman: A case report

作者：Jamal Hasoon , Amnon A. Berger , Ivan Urits , VwaireOrhurhu , Omar Viswanath, Musa Aner.

Beth Israel Deaconess Medical Center, Department of Anesthesia, Critical Care, and Pain Medicine, Harvard Medical School, Boston, MA, United States of America(美国，波士顿)

编译：沈霖杨敏   审校：郑学胜

Abstract

Tarlov cysts are extradural meningeal cysts with collections of cerebrospinal fluid within the nerve sheath. These cysts are uncommon but tend to present more often in women. Symptomatic Tarlov cysts can lead to a variety of neurologic symptoms and painful conditions, including chronic pelvic pain. There is no consensus regarding the best treatment for symptomatic cysts. Surgical management has high rates of complication, including chronic pain, but better long-term results for symptom and cyst resolution. We describe a patient who developed worsening pelvic pain and lumbar radiculopathy after surgical management of her Tarlov cysts. Medication failed to relieve the pain, as did a variety of other procedures, before the patient ultimately received significant pain relief from high-frequency spinal cord stimulation. This case may provide guidance for physicians when managing patients suffering from symptomatic Tarlov cysts, or worsening pain symptoms after surgical management of these cysts.

骶管囊肿是一种由于脑脊液在神经鞘内聚集引起的硬膜外囊肿。这类囊肿临床上相对不常见的，但往往更常见于妇女。症状性骶管囊肿可导致多种神经症状和疼痛状况，包括慢性盆底疼痛。对于症状性囊肿的最佳治疗，目前尚无共识。手术治疗有相对较高的并发症发生率，包括慢性疼痛，但对症状的缓解及囊肿的长期治疗效果更好。我们介绍一位在接受骶管囊肿手术治疗后盆底疼痛进行性加重以及出现腰椎神经根病的患者。在药物保守治疗及其他治疗方式无效的情况下，患者最终通过高频脊髓刺激中获得显著的疼痛缓解。这个病例可以为临床医生在治疗有症状的骶管囊肿或手术治疗后疼痛症状进一步加重的患者提供指导。

Case Description

The patient was a 66-year-old woman with a longstanding history of chronic pelvic pain secondary to multiple large Tarlov cysts affecting the S1-S4 nerve roots. She had attempted medication management with acetaminophen, NSAIDs, and neuropathic pain medications. She had also undergone a series of epidural steroid injections and a trial of superior hypogastric plexus blocks but obtain minimal relief with these. She ultimately had surgery for the removal of the Tarlov cysts as well as sacral lamina reconstruction (Fig. 1). However, the patient's surgery resulted in worsening of her chronic pelvic pain, and also produced new-onset back pain and lumbar radiculopathy down both legs. The patient reported constant debilitating back and pelvic pain with intermittent stabbing and burning pain in her legs. Her worsening symptoms were uncontrolled with continued medication management as well as a repeat series of epidural steroid injections.

患者是一位66岁女性，长期有慢性盆底疼痛病史，继发于累及S1-S4神经根的多发巨大骶管囊肿。她曾尝试用对乙酰氨基酚、非甾体抗炎药和神经性疼痛药物进行药物治疗。她还接受了一系列硬膜外类固醇注射和上腹部下丛阻滞的试验，但这些都取得极少程度的缓解。她最终接受了骶管囊肿切除和骶骨板重建手术（图1）。然而，手术后她的慢性骨盆疼痛恶化，也产生了新发的背痛和双腿的腰神经根病。病人存在持续的背部和骨盆疼痛伴随间歇性刺伤和腿部灼痛。通过持续的药物治疗以及一系列重复的硬膜外类固醇注射，她的症状没有得到控制。

Given that the patient's worsening symptoms were unrelieved with both medication management and interventional pain procedures, we tried high-frequency spinal cord stimulation for her worsening pain and new-onset radiculopathy.

考虑到患者症状在药物治疗和介入性疼痛治疗中都没有得到缓解，我们尝试高频脊髓刺激治疗其疼痛恶化和新发神经根病。

The patient was counseled regarding the risks and benefits of the procedure and elected to proceed. A spinal cord stimulator lead was introduced into the epidural space and advanced to the superior endplate of T8. A second lead was placed at the superior endplate of T9 (Fig. 2). The patient presented for follow-up after the procedure and reported significant improvement in her symptoms. She noted that the use of SCS had resulted in a 90% improvement of her back pain, a 95% improvement in her pelvic pain, and N50% improvement in herradiculopathy. Additionally, she reported she was much more active and was able to decrease her medication use with the pain relief she obtained from spinal cord stimulation.

告知病人相关风险和获益后，患者选择接受治疗。治疗过程中，将脊髓刺激器导线引入硬膜外腔，并推进到胸8的上终板。第二根导线被放置在胸9的上终板上（图2）。病人在手术后接受随访，并报告其症状有明显改善。她注意到使用脊神经电刺激后腰痛改善了90%，盆底疼痛改善了95%，神经根病改善了50%。此外，她本人说，她变得更加积极，同时能够减少药物的使用，并从脊髓电刺激中缓解了疼痛。

Discussion

There is no consensus on the optimal management of symptomatic Tarlov cysts. Percutaneous cyst drainage is a nonsurgical intervention has been used to treat this condition. This treatment is only temporary though, as cysts tend to gradually reform and symptoms recur. In addition to percutaneous drainage, one study has demonstrated that cyst aspiration with the placement of fibrin glue can prevent recurrence of the cysts. However, these patients are also at significant risk for postprocedural aseptic meningitis.

对于有症状的骶管囊肿的最佳治疗尚无共识。经皮囊肿穿刺引流术是一种非手术治疗方法，已被用于治疗这种情况。但这种治疗效果只是暂时的，因为囊肿会逐渐复原，症状会复发。除了经皮穿刺引流外，一项研究已经证明囊肿抽吸加纤维蛋白胶可以防止囊肿复发。然而，这些患者也存在术后无菌性脑膜炎的显著风险。

Surgical treatment of symptomatic cysts varies and can involve complete cyst removal with excision of the affected posterior root and ganglion, decompressive laminectomy, cyst wall resection, and cyst fenestration. The success and complication rates vary greatly by procedure. Again, there is no consensus regarding when surgical management for Tarlov cysts is warranted, though one study suggested that cysts larger than 1.5 cm with associated radicular pain or bowel/bladder dysfunction may benefit the most from surgical intervention.

有症状的骶管囊肿的外科治疗方法各不相同，可包括完全切除囊肿，切除病变的后根和神经节，椎板减压切除，囊肿壁切除，囊肿开窗术。手术成功率和并发症发生率因手术方式而异。同样，对于何时手术治疗骶管囊肿尚无共识，尽管一项研究表明，大于1.5 cm的囊肿伴有相关的神经根痛或肠/膀胱功能障碍可从手术干预中获益最大。

We would also like to comment on the success of spinal cord stimulation in this patient. SCS has been proven to be effective for treating intractable neuropathic pain such as lumbar radiculopathy and postlaminectomy syndrome. There is also growing evidence that SCS can even be helpful for treating debilitating chronic visceral pelvic pain. We believe this case is of importance as it describes the complicated management of patients with symptomatic Tarlov cysts who ultimately fail to respond to conservative therapy. We also describe the use of SCS in this patient and the benefit it can provide for patients who are suffering from severe radiculopathy after surgery as well as those with chronic pelvic pain (Fig. 3).

我们还想评论一下这个病人脊髓刺激治疗的成功。脊髓电刺激已被证明是一种有效的治疗难治性神经源性疼痛的方法，如腰神经根病和椎板切除术后综合征。也有越来越多的证据表明，脊髓电刺激治疗甚至可以帮助治疗慢性盆底疼痛。我们相信这个病例很重要，因为它描述了有症状的骶管囊肿患者的综合处理，而且这些患者对保守治疗无效。我们还描述了脊髓电刺激在该患者中的应用及其对术后严重神经根病以及慢性盆底疼痛患者的益处（图3）。

专家点评：郑学胜主任认为该患者是继发于骶管囊肿的慢性盆底疼痛，且本例患者骶管囊肿的治疗方式值得商榷。骶管囊肿的手术治疗核心在于封堵，而不是囊肿切除，切除的所谓“囊壁”是正常的神经根袖，将囊肿完全切除势必会损伤骶管内正常的神经，从而导致术后症状加重，甚至有大小便功能障碍的风险。临床上，已经遇到许多骶管囊肿的患者由于行骶管囊肿切除术导致不可逆的神经损伤，因而这种手术方式并不可取。此外，从术后磁共振上看，该患者切除了骶骨椎板，破坏骶骨的完整性，使得硬脊膜内巨大的静水压直接作用于腰骶部肌肉，这也是导致患者术后腰骶部疼痛重要原因之一。

该患者在手术治疗后症状进行性加重，最后采取了脊髓神经电刺激治疗缓解了疼痛症状。骶管囊肿可以引起腰骶部、会阴部、盆底、下肢疼痛，排尿排便功能障碍等症状，骶管囊肿的手术治疗仅仅是针对病因治疗，即使解除了病因，长期慢性的神经损伤会导致部分患者术后慢性盆底疼痛、排尿排便功能恢复不甚理想，而在药物、理疗、疼痛介入治疗等方式效果不理想的情况，可以建议此类患者考虑行神经电刺激治疗。目前神经电刺激治疗日趋成熟，包括脊髓电刺激、骶神经刺激对于改善慢性盆底疼痛均有良好的效果。此外，骶神经刺激还可以针对排尿排便功能恢复不理想的患者进行针对性的改善。临床上，已经有许多患者已经从神经电刺激治疗中获益。