SPINE Volume 39, Number 8, pp E538-E541 ©2014, Lippincott Williams & Wilkins

CASE REPORT

Symptomatic Spinal Cord Kinking Due to Focal Adhesive Arachnoiditis, With Ossification of the Ligamentum Flavum A Case Report Masashi Miyazaki, MD, PhD, Toyomi Yoshiiwa, MD, PhD, Toshinobu Ishihara, MD, Nobuhiro Kaku, MD, PhD, Masanori Kawano, MD, PhD, and Hiroshi Tsumura, MD, PhD

Study Design. A case report. Objective. To describe a rare case of symptomatic spinal cord kinking due to focal adhesive arachnoiditis, with ossification of the ligamentum flavum (OLF). Summary of Background Data. Spinal cord kinking without spinal surgery is rare, and symptomatic spinal cord kinking due to focal adhesive arachnoiditis, with OLF is even rarer. Methods. A 66-year-old female presented with numbness of the lower extremities and subsequently experienced gait disturbance due to motor weakness. Magnetic resonance imaging of the thoracic spine showed anterior displacement and kinking of the spinal cord from T11 to T12. Laminectomy and OLF resection were performed. The arachnoid membrane at the affected part was markedly thick and seemed cloudy. Adhesiolysis for arachnoid adhesion and release of spinal kinking were performed. Results. She could walk with a cane 6 months postoperatively. One year postoperatively, thoracic computed tomography–myelography showed that the cord was repositioned in the dural sac, and that release of the spinal cord kink was maintained. Conclusion. Symptomatic spinal cord kinking due to focal adhesive arachnoiditis, with OLF is a rare clinical condition. It was difficult to diagnose the precise pathology of the spinal cord before surgery. Microsurgical arachnoidolysis resolved the spinal cord kinking, and no recurrence was noted within the follow-up period.

From the Department of Orthopaedic Surgery, Oita University, Oita, Japan. Acknowledgment date: November 21, 2013. First revision date: December 21, 2013. Acceptance date: January 9, 2014. The manuscript submitted does not contain information about medical device(s)/drug(s). No funds were received in support of this work. No relevant financial activities outside the submitted work. Address correspondence and reprint requests to Masashi Miyazaki, MD, PhD, Department of Orthopaedic Surgery, Faculty of Medicine, Oita University, 1-1 Idaigaoka, Hasama-machi, Yufu-shi, Oita 879-5593, Japan; E-mail: [email protected] DOI: 10.1097/BRS.0000000000000225

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Key words: adhesive arachnoiditis, ossification of the ligamentum flavum, spinal cord kinking, paraplegia. Level of Evidence: N/A Spine 2014;39:E538–E541

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dvances in magnetic resonance imaging (MRI) technologies have enabled observation of various spinal cord morphologies before surgery. However, it may be difficult to diagnose the exact spinal cord pathologies in some cases. Spinal cord kinking is rare and is sometimes accompanied by idiopathic spinal cord herniation1 or spine shortening.2 Previously, hemorrhage in the cerebrospinal fluid or infections were considered a cause of adhesive arachnoiditis.3 However, in recent years, contrast media,4 trauma,5 epidural steroids,6 and local anesthesia7,8 have all been implicated as causes of adhesive arachnoiditis. Adhesive arachnoiditis with ossification of the ligamentum flavum (OLF) is rare,9 although the spinal cord kinking is not present. We present a case of symptomatic spinal cord kinking due to focal adhesive arachnoiditis with OLF.

CASE PRESENTATION A 66-year-old female presented with lower extremity numbness and subsequently experienced gait disturbance due to motor weakness after she slipped and experienced falls several times at home. She had no history of meningitis or previous spinal operations but had severe diabetes mellitus. The numbness and motor weakness in her lower extremities had deteriorated progressively for 3 months; therefore, she underwent medical examination. Neurological examination indicated diminished tactile and pinprick sensation, reduced temperature sensation below T12, severe bilateral lower extremity numbness, and diminished vibration and position sense. Patellar and Achilles tendon reflexes were hypotonic. She displayed muscle weakness in both legs, with 1 to 3 of 5 for strength in all key muscles. Although she had almost normal anal sensation and contraction, her urological examination

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CASE REPORT

Spinal Cord Kinking With Adhesive Arachnoiditis • Miyazaki et al

Figure 1. Thoracic spine MR image showing anterior displacement and spinal cord kinking at T11–T12. A, Sagittal-enhanced T1-weighted image showing no enhanced area in the spinal cord. B, Sagittal T2weighted image showing anterior displacement and spinal cord kinking at T11–T12 and high signal intensity area in the kinked cord, without enlarged dorsal subarachnoid spaces. C, Axial T2-weighted image at T11. MR image indicates magnetic resonance image.

revealed neurological bladder dysfunction. Thoracic spine MRI revealed anterior displacement and spinal cord kinking at T11–T12 and a high signal intensity area in the kinked cord on the sagittal and axial T2-weighted images, but no enlarged dorsal subarachnoid space. Moreover, enhanced MRI showed no enhanced area in the spinal cord (Figure 1A–C). Computed tomography–myelography revealed anterior angulation and undulation of the spinal cord at T11–T12 and OLF at intervertebral disc level of T11–T12 (Figure 2A–B). Spinal surgery was performed in the prone position under general anesthesia. Laminectomy from T10 to T12 and right facetectomy at T11–T12 were performed. OLF at the T11– T12 level compressed the dural sac from the lateral dorsal direction; therefore, ossifications were removed to obtain sufficient decompression. The dorsal dura mater was incised carefully at the midline; then, the microscopic observation of the arachnoid membrane showed that the affected part was markedly thick and cloudy (Figure 3). Arachnoid dissection was started on the dorsolateral side, and the adhered arachnoid membrane to the pia mater in the spinal cord was dissected as much as possible to mobilize the spinal cord. The dentate ligaments were relieved at both sides. At the ventral side, the spinal cord was adhered to the arachnoid membrane and tethered to the ventral side, although those were not the anterior dural defects or the herniated portions of the spinal cord. The arachnoid membrane adhering to the pia mater anteriorly was also dissected. To prevent readhesion, a 0.1-mm thick, expanded polytetrafluoroethylene pericardial membrane (GORE PRECLUDE; Flagstaff, AZ) was inserted and slid to the contralateral side. Afterward, both ends of the expanded polytetrafluoroethylene membrane were trimmed and sutured on the dural edge. The dorsal dura was approximated without duraplasty. Fibrin glue was used to prevent cerebrospinal fluid leakage. Finally, posterolateral fusion with spinal instrumentation was performed from T10 to T12. Neurological examination indicated gradual recovery of the preoperative motor weakness during the postoperative course. The patient’s tactile sensation was preserved in Spine

both legs, without any anal contraction. She was hospitalized for rehabilitation. She could walk with a cane at 6 months postoperatively. One year postoperatively, thoracic computed tomography–myelography showed that the cord was repositioned in the dural sac, and that the release of the spinal cord kinking was maintained (Figure 4 A–B).

DISCUSSION MRI is the “gold standard” for diagnosing spinal cord conditions. Spinal cord kinking is rare and may occur after spinal column shortening surgery for spinal deformity or spinal

Figure 2. CT myelography of the thoracic spine showing anterior angulation and undulation of the spinal cord from T11 to T12. A, Sagittal reconstruction image. B, Axial image showing OLF at T11–T12. CT indicates computed tomography; OLF, ossification of the ligamentum flavum. www.spinejournal.com

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CASE REPORT

Figure 3. An intraoperative picture showing the arachnoid membrane at the affected part was markedly thick and seemed cloudy. Adhesiolysis for arachnoid adhesion and release of spinal kinking were performed.

tumor. On sagittal MRI, typical features are ventral displacement and angulation of the spinal cord. The differential diagnosis is spinal cord herniation.10 In this case, ventral displacement and angulation of the spinal cord were detected on MRI. However, no enlarged dorsal subarachnoid space or spinal cord atrophy was observed. In addition, enhanced MRI showed no enhanced area in the spinal cord. Therefore, it would have been incorrect to consider a diagnosis of idiopathic spinal cord herniation or spinal cord tumor before surgery. However, the patient had not undergone any previous spinal operations and had not experienced vertebral

Spinal Cord Kinking With Adhesive Arachnoiditis • Miyazaki et al

compression fractures. The cause of spinal cord kinking was unknown before surgery. Kawahara et al11 recently reported the morphometric and physiological effects of acute spinal column shortening in a dog model. Their study describes spinal kinking in the gradual shortening of the spine. They stated that shortening of more than two-thirds of a single vertebral segment is characterized by spinal cord kinking and neurological dysfunction, and that shortening greater than two-thirds of the vertebrectomy length was in a “dangerous range.” They specified that excessive kinking results in neurological dysfunction such as morphological deformation of the cord, decreased spinal cord blood flow, and anterior spinal artery obstruction. This case showed a high signal intensity area in the kinked cord on the T2-weighted images and neurological dysfunction related to kinked spinal cord ischemia. This case had severe diabetes mellitus. It has been documented that the incidence of thoracic OLF is higher in the patients with diabetes mellitus.12 However, adhesive arachnoiditis with OLF is rare.9 Repeated minor mechanical damage of the spinal cord by the OLF could induce adhesive arachnoiditis and cause a tethering effect on the spinal cord. Studies have suggested that spinal tissue scarring at the injury site may cause tethering, which may significantly alter the spinal cord parenchyma.13 The morphometric changes in the spinal cord and the damage to the parenchyma may induce neurological deficit. Microsurgical decompression of the subarachnoid space has been reported for treating adhesive arachnoiditis.14 Although demanding, microsurgical arachnoidolysis is a straightforward method for mobilizing the spinal cord and reconstructing the cerebrospinal fluid pathway. In this case, an expanded polytetrafluoroethylene membrane was successfully used to prevent or minimize postoperative readhesion.

CONCLUSION Symptomatic spinal cord kinking due to focal adhesive arachnoiditis, with OLF is a rare clinical condition. Thoracic spinal MRI showed ventral kinking of the spinal cord; however, the precise pathology of the spinal cord was difficult to diagnose before surgery. Microsurgical arachnoidolysis resolved the spinal cord kinking, and no recurrence was noted within the follow-up period.

➢ Key Points

Figure 4. CT myelography of the thoracic spine 1 year after surgery showing that the cord was repositioned in the dural sac, and that the release of the spinal cord kinking was maintained. A, At sagittal reconstruction image, the spinal cord kinking was released and the dorsal root appeared at T12 level in expanded dorsal subarachnoid space. B, Axial image at T11. CT indicates computed tomography.

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‰ Symptomatic spinal cord kinking due to focal adhesive arachnoiditis, with OLF is a rare clinical condition. ‰ It was difficult to diagnose the precise pathology of the spinal cord before surgery. ‰ Microsurgical arachnoidolysis resolved the spinal cord kinking, and no recurrence was noted within the follow-up period.

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CASE REPORT References

1. Imagama S, Matsuyama Y, Sakai Y, et al. Image classification of idiopathic spinal cord herniation based on symptom severity and surgical outcome: a multicenter study. J Neurosurg Spine 2009;11:310–9. 2. Alemdaroglu KB, Atlihan D, Cimen O, et al. Morphometric effects of acute shortening of the spine: the kinking and the sliding of the cord, response of the spinal nerves. Eur Spine J 2007;16: 1451–7. 3. Rice I, Wee MY, Thomson K. Obstetric epidurals and chronic adhesive arachnoiditis. Br J Anaesth 2004;92:109–20. 4. Howell CJ, Dean T, Lucking L, et al. Randomised study of longterm outcome after epidural versus non-epidural analgesia during labour. BMJ 2002;325:357. 5. Kane RE. Neurologic deficits following epidural or spinal anesthesia. Anesth Analg 1981;60:150–61. 6. Abram SE, O’Connor TC. Complications associated with epidural steroid injections. Reg Anesth 1996;21:149–62. 7. Gemma M, Bricchi M, Grisoli M, et al. Neurologic symptoms after epidural anaesthesia. Report of three cases. Acta Anaesthesiol Scand 1994;38:742–3.

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Spinal Cord Kinking With Adhesive Arachnoiditis • Miyazaki et al

8. Killeen T, Kamat A, Walsh D, et al. Severe adhesive arachnoiditis resulting in progressive paraplegia following obstetric spinal anaesthesia: a case report and review. Anaesthesia 2012;67:1386–94. 9. Arai A, Aihara H, Miyake S, et al. Syringomyelia due to thoracic spinal stenosis with ossified ligamentum flavum—case report. Neurol Med Chir (Tokyo) 2011;51:157–9. 10. Brugieres P, Malapert D, Adle-Biassette H, et al. Idiopathic spinal cord herniation: value of MR phase-contrast imaging. AJNR Am J Neuroradiol 1999;20:935–9. 11. Kawahara N, Tomita K, Kobayashi T, et al. Influence of acute shortening on the spinal cord: an experimental study. Spine (Phila Pa 1976) 2005;30:613–20. 12. Li F, Chen Q, Xu K. Surgical treatment of 40 patients with thoracic ossification of the ligamentum flavum. J Neurosurg Spine 2006;4:191–7. 13. Morikawa T, Takami T, Tsuyuguchi N, et al. The role of spinal tissue scarring in the pathogenesis of progressive post-traumatic myelomalacia. Neurol Res 2006;28:802–6. 14. Ohata K, Gotoh T, Matsusaka Y, et al. Surgical management of syringomyelia associated with spinal adhesive arachnoiditis. J Clin Neurosci 2001;8:40–2.

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Symptomatic spinal cord kinking due to focal adhesive arachnoiditis, with ossification of the ligamentum flavum: a case report.

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