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Case report| Volume 38, 102126, March 2023

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Complete resolution of syrinx after circumferential adhesiolysis without deformity correction in a case of 6 year old neglected cervical facet dislocation

Published:February 11, 2023DOI:https://doi.org/10.1016/j.jcot.2023.102126

      Abstract

      Neglected traumatic cervical dislocation can be difficult to treat as such and more so if they are accompanied by an associated post traumatic syringomyelia (PTS). We present the case of a 55 year old gentleman who had a traumatic C6–C7 grade 2 listhesis which was neglected and presented 6 year later with 6 months history of neck pain, spastic quadriparesis and bowel bladder involvement. The patient was diagnosed with a PTS extending from C4 to D5. The possible aetiology and management of such cases has been discussed. The patient was successfully treated with decompression, adhesiolysis of arachnoid bands and syringotomy without the correction of deformity. The patient improved neurologically and had complete resolution of syrinx at final followup.

      Keywords

      1. Introduction

      Traumatic cervical facet dislocation may often be missed in developing countries, with a lot of patients not seeking medical attention especially if the patient starts improving neurologically. However, even after initial neurological improvement, an ongoing compression on the cord and change in alignment can lead to delayed syrinx formation. While, deformity correction with an aim of correcting the alignment is often recommended as treatment for these patients, we report a case where syrinx resolved and patient recovered neurologically in a 6 year old neglected C6-7 facet dislocation case managed by intradural procedure and circumferential arachnoid adhesiolysis without deformity correction.

      2. Case report

      A 55 year old male had a history of fall from height 6 years back in which he sustained blunt trauma to neck following which he developed quadriplegia. He didn't seek medical care for the same. However, he recovered completely over a period of 3 weeks. Six years later, he presented with complaints of neck pain and ascending spastic quadriparesis which was gradually progressive over 6 months associated with bowel and bladder involvement. On examination, he was found to have 4-/5 power in right upper limb, 3/5 in right lower limb, 4+/5 in left upper limb, 4/5 in left lower limb with increased tone in both lower limbs and extensor plantar responses. There was ∼30% sensory loss below C5 level.
      Radiographs of the cervical spine showed Grade II anterolisthesis of C6 over C7 vertebrae. No mobility was noted on flexion and extension views. MRI showed cord compression at C6-7 level and cervico-dorsal syrinx extending from C4 to D5 (Fig. 1). NCCT cervical spine showed solid bony fusion between the adjacent vertebral bodies and facets of C6– C7. It was fused in dislocated position (Fig. 2.)
      Fig. 1
      Fig. 1Preoperative computed tomography scans A) Right Parasagittal cut B) mid sagittal cut c) Left parasagittal cut showing grade 2 listhesis of C6 over C7 with solid fusion between vertebral bodies and facets in dislocated position.
      Fig. 2
      Fig. 2Preoperative sagittal T2 MRI showing cord compression at C6-7 level and cervico-dorsal syrinx extending from C4 to D5.
      The patient was planned for surgery and operated in prone position under general anaesthesia. A standard midline exposure was carried out and intraoperatively anterior displacement of C6 lamina over the C7 was seen. We performed a C6 and C7 laminectomy following which the hypertrophied ligamentum flavum which was densely adherent to duramater was carefully removed. A midline durotomy was done. On opening the dura, cord was expanded due to syringomyelia. It was decompressed by a midline syringotomy. Multiple arachnoid adhesions tethering the cord to duramater were seen circumferentially, which were divided. Dura was closed primarily. Immediate postoperative period was uneventful with no new neurological deficits. At 1 year followup, he showed significant improvement with diminution of neck pain and spasticity. MRI obtained at 1 year followup showed near total resolution of syrinx (Fig. 3).
      Fig. 3
      Fig. 3Postoperative sagittal T2 MRI at 1 year followup showing near total resolution of syrinx.

      3. Discussion

      Subaxial cervical spine is a common location for subluxations and dislocations.
      • Platzer P.
      • Hauswirth N.
      • Jaindl M.
      • Chatwani S.
      • Vecsei V.
      • Gaebler C.
      Delayed or missed diagnosis of cervical spine injuries.
      These are flexion -distraction injuries which may be associated with fractures of anterior or posterior elements. These cases are generally treated expeditiously to provide stability and prevent and mitigate sequelae such as prolonged pain, spinal cord and nerve injury, cervical deformity and syrinx formation. Delayed presentation of such cases is rare and ideal management of these cases is still not clear.
      • Srivastava S.K.
      • Aggarwal R.A.
      • Bhosale S.K.
      • Nemade P.S.
      Neglected dislocation in sub-axial cervical spine: case series and a suggested treatment protocol.
      Restoration of spine alignment is recommended in spine trauma even in patients with complete neurological deficits to prevent delayed syrinx formation which can threaten neurological function of proximal levels.
      Syringomyelia refers to cystic dilatation within the spinal cord and can be due to varied causes.
      • Milhorat T.H.
      • Capocelli A.L.
      • Anzil A.P.
      • Kotzen R.M.
      • Milhorat R.H.
      Pathological basis of spinal cord cavitation in syringomyelia: analysis of 105 autopsy cases.
      A study investigating patients with spinal cord injury at intervals ranging between 1 and 30 years post initial injury noted that nearly one-fourth of patients had a syrinx and some cystic spinal changes were noted in 30–50% of patients. Despite the high number of radiological changes only 1–9% patients had symptomatic syringomyelia.
      • Brodbelt A.R.
      • Stoodley M.A.
      Post-traumatic syringomyelia: a review.
      • Backe H.A.
      • Betz R.R.
      • Mesgarzadeh M.
      • Beck T.
      • Clancy M.
      Post-traumatic spinal cord cysts evaluated by magnetic resonance imaging.
      • Williams B.
      Pathogenesis of post-traumatic syringomyelia.
      • Wang D.
      • Bodley R.
      • Sett P.
      • Gardner B.
      • Frankel H.
      A clinical magnetic resonance imaging study of the traumatised spinal cord more than 20 years following injury.
      • Perrouin-Verbe B.
      • Lenne-Aurier K.
      • Robert R.
      • et al.
      Post-traumatic syringomyelia and post-traumatic spinal canal stenosis: a direct relationship: review of 75 patients with a spinal cord injury.
      • Abel R.
      • Gerner H.J.
      • Smit C.
      • Meiners T.
      Residual deformity of the spinal canal in patients with traumatic paraplegia and secondary changes of the spinal cord.
      The development of Post Traumatic Syrinomyelia (PTS) has been attributed to a number of factors like hematoma liquefaction, ischemia at the site of injury, autolysis and subarachnoid scaring which limit the flow of CSF.
      • Fairholm D.J.
      • Turnbull I.M.
      Microangiographic study of experimental spinal cord injuries.
      In PTS, a delayed progressive myelopathy often develops at spinal segments distant from the level of original lesion. Stenosis of spinal canal and kyphotic deformity have also been postulated as causes for development and progression of PTS.
      • Perrouin-Verbe B.
      • Lenne-Aurier K.
      • Robert R.
      • et al.
      Post-traumatic syringomyelia and post-traumatic spinal canal stenosis: a direct relationship: review of 75 patients with a spinal cord injury.
      ,
      • Abel R.
      • Gerner H.J.
      • Smit C.
      • Meiners T.
      Residual deformity of the spinal canal in patients with traumatic paraplegia and secondary changes of the spinal cord.
      Post traumatic syrinx is a surgical challenge and its management remains controversial. The identification of the underlying cause and its correction remain the main goals of surgical management. The various surgical options include deformity correction, decompression, shunting procedures, arachnoid adhesiolysis with or without duroplasty, and transection of the cord in dire circumstances. Regarding the preferred therapeutic strategies, in cases of motor neurological deficit, a surgical intervention involving spinal cord adhesiolysis with expansive duroplasty has been recommended as the preferred first-line surgical technique.
      • Bonfield C.M.
      • Levi A.D.
      • Arnold P.M.
      • Okonkwo D.O.
      Surgical management of post-traumatic syringomyelia.
      Kliendienst et al. in their review article on 866 PTS patients noted arachnoid lysis (48%) and drainage of syrinx by various techniques (31%) as the most commonly used techniques. Less frequently used procedures included cord transection, syringostomy, duroplasty, a combination of arachnoid lysis syringo-subarachnoid shunt and decompression alone.
      • Kleindienst A.
      • Laut F.M.
      • Roeckelein V.
      • Buchfelder M.
      • Dodoo-Schittko F.
      Treatment of posttraumatic syringomyelia: evidence from a systematic review.
      However, shunting is associated with problems of shunt failure with about half of them becoming non-functional at 6 years.
      • Sgouros S.
      • Williams B.
      A critical appraisal of drainage in syringomyelia.
      Besides the syrinx formation, the ongoing compression of the cord and kyphotic alignment are important factors contributing to neurological deterioration. In such cases decompression, realignment and stabilization of spine become equally important.
      • Schurch B.
      • Wichmann W.
      • Rossier A.B.
      Post-traumatic syringomyelia (cystic myelopathy): a prospective study of 449 patients with spinal cord injury.
      In the present case, the patient had a grade 2 listhesis of cervical body, however a solid fusion was seen anteriorly as well as posteriorly. Moreover, the alignment of the spine was straight. The focal compression and straight/lordotic curvature made us go for arachnoid adhesiolysis and syringotomy alone instead of adding deformity correction also. The latter would have resulted in significantly higher operative time, cost, blood loss and morbidity for the patient. Moreover, deformity correction and fixation would have compromised the motion of included segments and also would have increased the risk of adjacent segment disease. If patient had come with kyphosis at index level a more extensive procedure of posterior release of facets followed by anterior corpectomy and restoration of lordosis probably would have been necessary. Laminectomy and circumferential adhesiolysis restored CSF circulation and lead to disappearance of syrinx with neurological improvement.

      4. Conclusion

      Syrinx due to focal compression in cervical spine when curvature is lordotic can be treated by laminectomy and circumferential release of cord without bony deformity correction.

      Funding statement

      There is no funding source for this publication/study.

      Author statement

      Hitesh Gurjar (H.G): Concept and Design, Data Analysis, Manuscript preparation, Manuscript Editing, Approval of final version of manuscript.Abhilash Reddy D (A.R.D): Concept and Design, Data Acquistion, Data Analysis, Manuscript preparation, Manuscript Editing, Approval of final version of manuscript.Tungish Bansal(T.B): Concept and Design, Data Acquistion, Data Analysis, Manuscript preparation, Manuscript Editing, Approval of final version of manuscript.

      Declaration of competing interest

      Dr.Hitesh Gurjar, Dr. Abhilash Reddy D, Dr. Tungish Bansal declare that they have no conflict of interest.

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