Management of Neglected Post-traumatic C4/C5 Spondyloptosis in a Low Resource Setting: a Review of Literature and Case Study.

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Christopher Anele, Chizowa Ezeaku, Simon Balogun, Temitope Ajekwu, and 2 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-3821132/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Objectives To highlight the incidence, outcome, and peculiar management challenges of cervical spondyloptosis Methods A retrospective literature review and case study Results Five cases of subaxial cervical spondyloptosis were identified from the literature and reviewed. Their demographics, mechanisms of trauma, intervention, and outcome were highlighted. Conclusion Subaxial cervical spondyloptosis is relatively uncommonly documented in the literature. The management outcomes are variable, and peculiar management challenges are highlighted in the index case study. Neurosurgery Cervical spondyloptosis Neglected Anterior-approach Figures Figure 1 Figure 2 Figure 3 Figure 4 Introduction Post-traumatic cervical spondyloptosis (PTCS) is an uncommon form of cervical spine injury with total slippage of the vertebral body over the subjacent (> 100%). Although there are reported cases with intact neurology, it is usually associated with significant neurological impairment and mortality. 1 , 2 As of 2022, there are about 66 cases documented worldwide, which may be attributed to the selectivity of reporting cases with favourable outcomes, rather than their rarity. 3 While there is no consensus definition, neglected PTCS has been described as a delay in instituting standard management in patients with PTCS, especially when it extends beyond 3–4 weeks from trauma. 4 , 5 Its causes are usually multifactorial and are associated with longer hospital stays, complex surgical procedures, complications, and poor neurological outcomes. 6 We present a case of neglected C4/C5 spondyloptosis managed with improved neurologic outcomes, highlighting the peculiar management challenges. We also reviewed similar cases of the same cervical level reported in the literature. Case Study A 50-year-old right-handed female presented to our clinic with an eight-month history of posterior neck pain and weakness of all limbs following a fall in the bathroom. This was associated with paraesthesia on the upper extremities and bi-sphincteric dysfunction in favour of constipation and urinary retention. Four months into illness, she began to have spasms involving the trunk and all extremities. There were no intracranial symptoms nor trauma to other parts of the body. She has been managed in the past for pulmonary tuberculosis. She initially sought traditional care and defaulted from private specialist care due to cost, before presenting to our neurosurgical outpatient clinic. She was fully conscious but in painful distress, had a Philadelphia cervical collar and sat supported on a wheelchair. She had gross muscle wasting, hyperreflexia (deep tendon grade 4) and hypertonia on all extremities. Her motor level was C6, and her sensory level was C7 with a preserved sacral segment. She also had flexor contractures on both knees. Magnetic resonance imaging, MRI, (Fig. 1 ) done at the presentation showed grade five anterolisthesis of the C4/C5 with devitalised C5 and C6 body and adjacent cord thinning. A chest x-ray showed left lobar collapse with compensatory emphysema on the right. A diagnosis of neglected C6 quadriplegia, AIS C with C4/C5 spondyloptosis (AO spine classification C4 Type C, A0, N3). She was placed on a muscle relaxant and spasmolytics. In addition, Gardner well traction was applied for 2 weeks. An anterior cervical corpectomies of C5 and C6 was done, with interbody fusion with an 18mm expandable titanium cage, a 6-hole anterior plate and four 16mm-screws bridging C4-C7. Intraoperative findings were of florid anterior marginal osteophytes, coronal malalignment, abnormal and devitalised C5 and C6 bodies with pseudoarthrosis, healed C6 body fracture, and reduced C4/C5 and C6/C7 discs. Intraoperative fluoroscopic images (Fig. 3 ) showed good reduction and alignment. The post-operative period was uneventful and steady neurological improvement was noted. On the 4-week follow-up, the bi-sphincteric dysfunction was resolved, and she could sit without support, the motor function improved to AIS-D, with a resolution of truncal and limb spasms. A cervical spine x-ray (Fig. 4) confirmed good alignment with intact hardware. She is presently undergoing physiotherapy and has been planned for bilateral hamstring tendons lengthening on account of contractures. Summary of Reviewed Cases in Literature with C4/C5 spondyloptosis References Age/Sex Mechanism of Injury Time to presentation Pre op AIS Treatment Modalities Post op AIS Tumialan and Theodore 7 (2013) 43 years/ Male MVA Immediate A Traction + Anterior approach Died, 24 hours post-op Gascol et al. 8 (2013) 45 years/ Male MVA 8 months E Traction + combined approach (540°) ‘E’ immediate post-op Özdoğan et al. 1 (2017) 36 years/ Female Fell from tree Immediate A Anterior approach Not clearly stated (Minimal improvement) Saleh et al. 9 (2017) 2 weeks/ Male Birth trauma 2 weeks C Traction (failed) + Combined approach ‘E’ at 4.5 years Rokaya et al. 10 (2020) 22 years/ Male Injury to the neck from a falling tree Immediate A Traction + combined approach ‘A’ at 1 month Our case 50 years/ Female Slippage and fall on the bathroom 8 months C Traction + anterior approach ‘D’ at 4 weeks KEY: MVA = Motor vehicular accident, op = Operative, AIS = American Spinal Injury Association Impairment Scale Discussion Posttraumatic cervical spondyloptosis is usually caused by a high impact force. Road traffic accidents are the most common mechanism of injury. However, unusual mechanisms such as vaginal delivery, intubation, spinal massage or manipulation, and indoor slippage and fall, as in our index case, have been reported. 9 , 11 – 13 Available data show that the C7-T1 level was the most affected segment. 3 , 14 Six cases of C4-C5 level involvement, similar to our index case, have been reported and are summarized in Table 1. The time of presentation from trauma ranges from a few hours to eight years. 2 , 3 , 9 , 13 , 14 Interestingly, our patient and those reported by Gascol et al. 8 , Sakta et al. 13 , and a case reported by Padwal et al. 15 , all presented eight months following trauma. Neglected PTCS poses numerous management challenges. These include complications from prolonged immobilisation such as pressure sores, recurrent urinary tract infections, constipation, and joint contractures, as in the present case. 14 They are also at an increased risk of progressive neurological deterioration, intraoperative blood loss, damage to neural structures, and residual spinal deformity. This results in a prolonged hospital stay, increased costs, and poor outcomes. 6 The paucity of specialist facilities, overlooked or missed diagnoses, ignorance, poverty, and unregulated proliferation of traditional healers have been implicated in delayed presentation. 5 , 13 This is coupled with locoregional challenges such as poor health-seeking attitudes, limited health insurance coverage, reduced manpower from brain drain, limited theatre spaces, and state-of-the-art neurosurgical facilities. 16 Our index case initially sought care with a traditional healer before presenting to our facility, thereby exhausting their limited funds before presenting in our facility. Occasionally, unconventional practices in the hands of this shamanist have been noted to worsen a patient's neurological status. 13 At present, there is no consensus in management. Various approaches have been described and tailored to patient presentation and neurologic deficit, neuroimaging findings, and surgeon preference. An all-out non-operative approach has been tried, but the outcome appears poor, with a high conversion rate to open reduction, and mortality reported in one. 3 , 17 In our index case, preoperative Gardner well traction was instituted to achieve some degree of reduction before surgery, and ameliorate the associated paraspinal spasms. This was achieved with a reduction to grade one listhesis (Fig. 2). Our observation is in contrast with a previous report 8 and recommendation 13 on the non-effectiveness of preoperative cervical traction in neglected cervical spondyloptosis. There is presently no well-designed study to evaluate the outcomes of various approaches. Initially, surgical reduction was thought to be unsafe, and at present, combined anterior and posterior approaches appear to be frequently performed, with proponents advocating better stabilization and cord decompression. 12 , 13 , 17 , 18 In our case, an anterior-only approach was chosen to address the marked anterior cord compression and excise the deformed and osteopenic C5 and C6 vertebrae bodies with pseudoarthrosis, to enable adequate decompression and instrumented fusion. A similar application of this procedure has been reported with an excellent outcome. 13 Also, anterior fusion stabilises two columns (anterior and middle Dennis column), especially if a bicortical purchase is achieved (see Fig. 4), like in our index case. 17 We also considered a shorter operative time due to her reduced lung capacity from previous pulmonary tuberculosis, which a single approach may guarantee compared to a combined approach. The posterior-only approach, the least frequently used option is suited for cases of persistent bilateral facet lock, following failed closed reduction, multi-level cervical fractures and to mitigate the risk of graft migration that could complicate anterior fusion. 13 , 19 , 20 Her postoperative period was unremarkable, but the following complications have been reported: CSF leak from dural tear and life-threatening vertebral or basilar artery injury, emphasising the need for perioperative CT angiography in the management of this patient. 7 , 14 The outcomes of cervical spondyloptosis are usually noted to be poor; however, within the limits of reported cases, neurological improvement was observed in 40%, with a mortality of 11% recorded. 3,21 Our index case had resolution of autonomic dysfunction with motor improvement from AIS C to D as of the 4th week post-operation. We are optimistic about improved motor activity with continued physiotherapy and the surgical release of both knee contractures. Conclusion Neglected post traumatic cervical spondyloptosis is a rare and lethal form of high-grade cervical injury with varied mechanism of injuries and modalities of treatment. While there is a need for studies with high level of evidence, at present, treatment should be individualised. Measure are needed to address the causes of late presentation with its attendant sequalae. Declarations Funding The authors received no external funding for the publication of the work. Consent A written informed consent was obtained from the patient for permission to publish the case with the accompanying images. This could be made to the editor-in-chief on request. Declaration of competing interest The authors have no competing interests. References Özdoğan S, Kaya M, Demirel N, Düzkalır AH, Yaltırık CK. Isolated C5 vertebrae dislocation with trauma: An extremely rare case of isolated C5 dislocation. Am J Case Rep. 2017 Nov 28;18:1256–60. Ramieri A, Domenicucci M, Cellocco P, Lenzi J, Dugoni DE, Costanzo G. Traumatic spondylolisthesis and spondyloptosis of the subaxial cervical spine without neurological deficits: Closed re-alignment, Surgical options and literature review. Eur Spine J. 2014 Nov 1;23:S658–63. Khelifa A, Berchiche L, Aichaoui F, Lagha N, Asfirane N, Morsli A. Traumatic cervical spine spondyloptosis: A systematic review. Vol. 13, Journal of Craniovertebral Junction and Spine. Wolters Kluwer -- Medknow Publications; 2022. p. 9–16. Bechet FR, Stassen P, Scorpie D, Della Siega T. Delayed Treatment of Traumatic Cervical Dislocation: A Case Report and Literature Review. Case Rep Orthop. 2022 Mar 2;2022:1–6. Ifthekar S, Ahuja K, Mittal S, Sarkar B, Deep G, Thomas W, et al. Management of Neglected Upper Cervical Spine Injuries. Indian J Orthop. 2021 Jun 1;55(3):673–9. Chhabra HS, Arora M. Neglected traumatic spinal cord injuries: Causes, consequences and outcomes in an Indian setting. Spinal Cord. 2013 Mar 1;51(3):238–44. Tumialán LM, Theodore N. Basilar artery thrombosis after reduction of cervical spondyloptosis: A cautionary report - Case report. J Neurosurg Spine. 2012 May;16(5):492–6. Gasco J, Dilorenzo DJ, Patterson JT. C4-C5 post-traumatic spondyloptosis with in situ fusion: Systematic literature review and case report. Spine (Phila Pa 1976). 2013 May 1;38(10). Saleh S, Swanson KI, Bragg T. Successful surgical repair and recovery in a 2-week-old infant after birth-related cervical fracture dislocation. J Neurosurg Pediatr. 2018 Jan 1;21(1):16–20. Rokaya PK, Khadka NK, Giri PK, Khapung R, Mahaseth N. Burst fracture of c5 with traumatic anterior spondyloptosis of c6 and posterior spondylolisthesis of c4 vertebra: A case report. J Nepal Med Assoc. 2021 Apr 1;59(236):402–5. Oppenlander ME, Hsu FD, Bolton P, Theodore N. Catastrophic neurological complications of emergent endotracheal intubation: Report of 2 cases. J Neurosurg Spine. 2015 May 1;22(5):454–8. Wong K, Chang P, Monasky M, Samuelson R. Traumatic spondyloptosis of the cervical spine: A case report and discussion of worldwide treatment trends. Surg Neurol Int. 2017;8(1). Sakti YM, Anzhari S, Kartika A, Irfantian A, Ahmad H, Sakadewa GP, et al. Neglected cervical spondyloptosis of the 5TH – 6TH cervical spine following cervical manipulation: A case report. Int J Surg Case Rep. 2022 May 1;94:106984. Modi JV, Soman SM, Dalal S. Traumatic cervical spondyloptosis of the subaxial cervical spine: A case series with a literature review and a new classification. Asian Spine J. 2016;10(6):1058–64. Padwal A, Shukla D, Bhat DI, Somanna S, Devi BI. Post-traumatic cervical spondyloptosis: A rare entity with multiple management options. J Clin Neurosci. 2016 Jun 1;28:61–6. Rehman AU, Ahmed A, Zaheer Z, Ahmed B, Lucke-Wold B. International Neurosurgery: The Role for Collaboration. Int J Med Pharm Res. 2023;4(1):15–24. Okoro EU, Havryliv TS, Smolanka AV SV. “Traumatic Cervical Anterior Spondyloptosis: Literature Review and Case Report.” SVOA Neurol. 2021;2(3):84–94. Bhojraj SY, Shahane SM. Posttraumatic cervical spondyloptosis at C6-7 with late-onset cord compression: A new clinical entity. Case report. J Neurosurg. 1992;77(5):792–4. Chadha M, Singh AP SA. Spondyloptosis of C6-C7: a rare case report. Chin J Traumatol. 2010;13(6):377–9. Ozdogan C, Gogusgeren MA DM. Posttraumatic cervical spondyloptosis “Case Report.” Turk J Trauma Emerg Surg. 1999;5:46–8. Ng C, Feldstein E, Spirollari E, Vazquez S, Naftchi A, Graifman G, et al. Management and outcomes of adult traumatic cervical spondyloptosis: A case report and systematic review. Vol. 103, Journal of Clinical Neuroscience. J Clin Neurosci; 2022. p. 34–40. Additional Declarations The authors declare no competing interests. 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imaging\u003c/p\u003e","description":"","filename":"3.png","url":"https://assets-eu.researchsquare.com/files/rs-3821132/v1/9d37c8266aed1ead84edb6fe.png"},{"id":49060807,"identity":"1ba22b6c-3abe-46b5-aaf3-7f0bda346b3b","added_by":"auto","created_at":"2024-01-02 14:10:03","extension":"jpeg","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":61157,"visible":true,"origin":"","legend":"\u003cp\u003eC-spine x-ray 4 weeks after surgery\u003c/p\u003e","description":"","filename":"image4.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-3821132/v1/822e5080bc2b10663b328e0d.jpeg"},{"id":49061601,"identity":"8871421c-2cf8-4d67-b575-bed0bc287d41","added_by":"auto","created_at":"2024-01-02 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Although there are reported cases with intact neurology, it is usually associated with significant neurological impairment and mortality.\u003csup\u003e\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e,\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e\u003c/sup\u003e As of 2022, there are about 66 cases documented worldwide, which may be attributed to the selectivity of reporting cases with favourable outcomes, rather than their rarity.\u003csup\u003e\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u003c/sup\u003e While there is no consensus definition, neglected PTCS has been described as a delay in instituting standard management in patients with PTCS, especially when it extends beyond 3\u0026ndash;4 weeks from trauma.\u003csup\u003e\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e,\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e\u003c/sup\u003e Its causes are usually multifactorial and are associated with longer hospital stays, complex surgical procedures, complications, and poor neurological outcomes.\u003csup\u003e\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e\u003c/sup\u003e We present a case of neglected C4/C5 spondyloptosis managed with improved neurologic outcomes, highlighting the peculiar management challenges. We also reviewed similar cases of the same cervical level reported in the literature.\u003c/p\u003e"},{"header":"Case Study","content":"\u003cp\u003eA 50-year-old right-handed female presented to our clinic with an eight-month history of posterior neck pain and weakness of all limbs following a fall in the bathroom. This was associated with paraesthesia on the upper extremities and bi-sphincteric dysfunction in favour of constipation and urinary retention. Four months into illness, she began to have spasms involving the trunk and all extremities. There were no intracranial symptoms nor trauma to other parts of the body. She has been managed in the past for pulmonary tuberculosis. She initially sought traditional care and defaulted from private specialist care due to cost, before presenting to our neurosurgical outpatient clinic.\u003c/p\u003e\n\u003cp\u003eShe was fully conscious but in painful distress, had a Philadelphia cervical collar and sat supported on a wheelchair. She had gross muscle wasting, hyperreflexia (deep tendon grade 4) and hypertonia on all extremities. Her motor level was C6, and her sensory level was C7 with a preserved sacral segment. She also had flexor contractures on both knees. Magnetic resonance imaging, MRI, (Fig. \u003cspan class=\"InternalRef\"\u003e1\u003c/span\u003e) done at the presentation showed grade five anterolisthesis of the C4/C5 with devitalised C5 and C6 body and adjacent cord thinning. A chest x-ray showed left lobar collapse with compensatory emphysema on the right.\u003c/p\u003e\n\u003cp\u003eA diagnosis of neglected C6 quadriplegia, AIS C with C4/C5 spondyloptosis (AO spine classification C4 Type C, A0, N3). She was placed on a muscle relaxant and spasmolytics. In addition, Gardner well traction was applied for 2 weeks. An anterior cervical corpectomies of C5 and C6 was done, with interbody fusion with an 18mm expandable titanium cage, a 6-hole anterior plate and four 16mm-screws bridging C4-C7. Intraoperative findings were of florid anterior marginal osteophytes, coronal malalignment, abnormal and devitalised C5 and C6 bodies with pseudoarthrosis, healed C6 body fracture, and reduced C4/C5 and C6/C7 discs. Intraoperative fluoroscopic images (Fig. \u003cspan class=\"InternalRef\"\u003e3\u003c/span\u003e) showed good reduction and alignment. The post-operative period was uneventful and steady neurological improvement was noted.\u003c/p\u003e\n\u003cp\u003eOn the 4-week follow-up, the bi-sphincteric dysfunction was resolved, and she could sit without support, the motor function improved to AIS-D, with a resolution of truncal and limb spasms. A cervical spine x-ray (Fig. 4) confirmed good alignment with intact hardware. She is presently undergoing physiotherapy and has been planned for bilateral hamstring tendons lengthening on account of contractures.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eSummary of \u0026nbsp;Reviewed Cases in Literature with C4/C5 spondyloptosis\u003c/strong\u003e\u003c/p\u003e\n\u003cdiv class=\"gridtable\"\u003e\u003ctable id=\"Taba\" border=\"1\"\u003e\n \u003ccolgroup cols=\"7\"\u003e\u003c/colgroup\u003e\n \u003cthead\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eReferences\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eAge/Sex\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eMechanism of Injury\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eTime to presentation\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003ePre op AIS\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eTreatment Modalities\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003ePost op AIS\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003c/thead\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTumialan and Theodore\u003csup\u003e\u003cspan class=\"CitationRef\"\u003e7\u003c/span\u003e\u003c/sup\u003e (2013)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e43 years/ Male\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eMVA\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eImmediate\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eA\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTraction\u0026thinsp;+\u0026thinsp;Anterior approach\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eDied, 24 hours post-op\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eGascol et al.\u003csup\u003e\u003cspan class=\"CitationRef\"\u003e8\u003c/span\u003e\u003c/sup\u003e (2013)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e45 years/\u003c/p\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eMVA\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e8 months\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eE\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTraction\u0026thinsp;+\u0026thinsp;combined approach (540\u0026deg;)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e\u0026lsquo;E\u0026rsquo; immediate post-op\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e\u0026Ouml;zdoğan et al.\u003csup\u003e\u003cspan class=\"CitationRef\"\u003e1\u003c/span\u003e\u003c/sup\u003e (2017)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e36 years/\u003c/p\u003e\n \u003cp\u003eFemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eFell from tree\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eImmediate\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eA\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eAnterior approach\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eNot clearly stated (Minimal improvement)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eSaleh et al.\u003csup\u003e\u003cspan class=\"CitationRef\"\u003e9\u003c/span\u003e\u003c/sup\u003e (2017)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e2 weeks/\u003c/p\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eBirth trauma\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e2 weeks\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eC\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTraction (failed)\u0026thinsp;+\u0026thinsp;Combined approach\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e\u0026lsquo;E\u0026rsquo; at 4.5 years\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eRokaya et al.\u003csup\u003e\u003cspan class=\"CitationRef\"\u003e10\u003c/span\u003e\u003c/sup\u003e (2020)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e22 years/\u003c/p\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eInjury to the neck from a falling tree\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eImmediate\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eA\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTraction\u0026thinsp;+\u0026thinsp;combined approach\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e\u0026lsquo;A\u0026rsquo; at 1 month\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eOur case\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e50 years/ Female\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eSlippage and fall on the bathroom\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e8 months\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eC\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTraction\u0026thinsp;+\u0026thinsp;anterior approach\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e\u0026lsquo;D\u0026rsquo; at 4 weeks\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n \u003ctfoot\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"7\"\u003eKEY: MVA\u0026thinsp;=\u0026thinsp;Motor vehicular accident, op\u0026thinsp;=\u0026thinsp;Operative, AIS\u0026thinsp;=\u0026thinsp;American Spinal Injury Association Impairment Scale\u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tfoot\u003e\n \u003c/table\u003e\n\u003c/div\u003e"},{"header":"Discussion","content":"\u003cp\u003ePosttraumatic cervical spondyloptosis is usually caused by a high impact force. Road traffic accidents are the most common mechanism of injury. However, unusual mechanisms such as vaginal delivery, intubation, spinal massage or manipulation, and indoor slippage and fall, as in our index case, have been reported.\u003csup\u003e\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e,\u003cspan additionalcitationids=\"CR12\" citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e Available data show that the C7-T1 level was the most affected segment.\u003csup\u003e\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e,\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e\u003c/sup\u003e Six cases of C4-C5 level involvement, similar to our index case, have been reported and are summarized in Table\u0026nbsp;1. The time of presentation from trauma ranges from a few hours to eight years.\u003csup\u003e\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e,\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e,\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e,\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e,\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e\u003c/sup\u003e Interestingly, our patient and those reported by Gascol et al.\u003csup\u003e\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e\u003c/sup\u003e, Sakta et al.\u003csup\u003e\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e, and a case reported by Padwal et al.\u003csup\u003e\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e\u003c/sup\u003e, all presented eight months following trauma.\u003c/p\u003e \u003cp\u003eNeglected PTCS poses numerous management challenges. These include complications from prolonged immobilisation such as pressure sores, recurrent urinary tract infections, constipation, and joint contractures, as in the present case.\u003csup\u003e\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e\u003c/sup\u003e They are also at an increased risk of progressive neurological deterioration, intraoperative blood loss, damage to neural structures, and residual spinal deformity. This results in a prolonged hospital stay, increased costs, and poor outcomes.\u003csup\u003e\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e\u003c/sup\u003e The paucity of specialist facilities, overlooked or missed diagnoses, ignorance, poverty, and unregulated proliferation of traditional healers have been implicated in delayed presentation.\u003csup\u003e\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e,\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e This is coupled with locoregional challenges such as poor health-seeking attitudes, limited health insurance coverage, reduced manpower from brain drain, limited theatre spaces, and state-of-the-art neurosurgical facilities.\u003csup\u003e\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e\u003c/sup\u003e Our index case initially sought care with a traditional healer before presenting to our facility, thereby exhausting their limited funds before presenting in our facility. Occasionally, unconventional practices in the hands of this shamanist have been noted to worsen a patient's neurological status.\u003csup\u003e\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eAt present, there is no consensus in management. Various approaches have been described and tailored to patient presentation and neurologic deficit, neuroimaging findings, and surgeon preference. An all-out non-operative approach has been tried, but the outcome appears poor, with a high conversion rate to open reduction, and mortality reported in one.\u003csup\u003e\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e,\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e\u003c/sup\u003e In our index case, preoperative Gardner well traction was instituted to achieve some degree of reduction before surgery, and ameliorate the associated paraspinal spasms. This was achieved with a reduction to grade one listhesis (Fig.\u0026nbsp;2). Our observation is in contrast with a previous report\u003csup\u003e\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e\u003c/sup\u003e and recommendation\u003csup\u003e\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e on the non-effectiveness of preoperative cervical traction in neglected cervical spondyloptosis.\u003c/p\u003e \u003cp\u003eThere is presently no well-designed study to evaluate the outcomes of various approaches. Initially, surgical reduction was thought to be unsafe, and at present, combined anterior and posterior approaches appear to be frequently performed, with proponents advocating better stabilization and cord decompression.\u003csup\u003e\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e,\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e,\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e,\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e\u003c/sup\u003e In our case, an anterior-only approach was chosen to address the marked anterior cord compression and excise the deformed and osteopenic C5 and C6 vertebrae bodies with pseudoarthrosis, to enable adequate decompression and instrumented fusion. A similar application of this procedure has been reported with an excellent outcome.\u003csup\u003e\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e Also, anterior fusion stabilises two columns (anterior and middle Dennis column), especially if a bicortical purchase is achieved (see Fig.\u0026nbsp;4), like in our index case.\u003csup\u003e\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e\u003c/sup\u003e We also considered a shorter operative time due to her reduced lung capacity from previous pulmonary tuberculosis, which a single approach may guarantee compared to a combined approach. The posterior-only approach, the least frequently used option is suited for cases of persistent bilateral facet lock, following failed closed reduction, multi-level cervical fractures and to mitigate the risk of graft migration that could complicate anterior fusion.\u003csup\u003e\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e,\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e,\u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eHer postoperative period was unremarkable, but the following complications have been reported: CSF leak from dural tear and life-threatening vertebral or basilar artery injury, emphasising the need for perioperative CT angiography in the management of this patient.\u003csup\u003e\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e,\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e\u003c/sup\u003e The outcomes of cervical spondyloptosis are usually noted to be poor; however, within the limits of reported cases, neurological improvement was observed in 40%, with a mortality of 11% recorded.\u003csup\u003e3,21\u003c/sup\u003e Our index case had resolution of autonomic dysfunction with motor improvement from AIS C to D as of the 4th week post-operation. We are optimistic about improved motor activity with continued physiotherapy and the surgical release of both knee contractures.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eNeglected post traumatic cervical spondyloptosis is a rare and lethal form of high-grade cervical injury with varied mechanism of injuries and modalities of treatment. While there is a need for studies with high level of evidence, at present, treatment should be individualised. Measure are needed to address the causes of late presentation with its attendant sequalae.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors received no external funding for the publication of the work.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eA written informed consent was obtained from the patient for permission to publish the case with the accompanying images. This could be made to the editor-in-chief on request.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eDeclaration of competing interest\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors have no competing interests.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003e\u0026Ouml;zdoğan S, Kaya M, Demirel N, D\u0026uuml;zkalır AH, Yaltırık CK. Isolated C5 vertebrae dislocation with trauma: An extremely rare case of isolated C5 dislocation. Am J Case Rep. 2017 Nov 28;18:1256\u0026ndash;60. \u003c/li\u003e\n\u003cli\u003eRamieri A, Domenicucci M, Cellocco P, Lenzi J, Dugoni DE, Costanzo G. Traumatic spondylolisthesis and spondyloptosis of the subaxial cervical spine without neurological deficits: Closed re-alignment, Surgical options and literature review. Eur Spine J. 2014 Nov 1;23:S658\u0026ndash;63. \u003c/li\u003e\n\u003cli\u003eKhelifa A, Berchiche L, Aichaoui F, Lagha N, Asfirane N, Morsli A. Traumatic cervical spine spondyloptosis: A systematic review. Vol. 13, Journal of Craniovertebral Junction and Spine. Wolters Kluwer -- Medknow Publications; 2022. p. 9\u0026ndash;16. \u003c/li\u003e\n\u003cli\u003eBechet FR, Stassen P, Scorpie D, Della Siega T. Delayed Treatment of Traumatic Cervical Dislocation: A Case Report and Literature Review. Case Rep Orthop. 2022 Mar 2;2022:1\u0026ndash;6. \u003c/li\u003e\n\u003cli\u003eIfthekar S, Ahuja K, Mittal S, Sarkar B, Deep G, Thomas W, et al. Management of Neglected Upper Cervical Spine Injuries. Indian J Orthop. 2021 Jun 1;55(3):673\u0026ndash;9. \u003c/li\u003e\n\u003cli\u003eChhabra HS, Arora M. Neglected traumatic spinal cord injuries: Causes, consequences and outcomes in an Indian setting. Spinal Cord. 2013 Mar 1;51(3):238\u0026ndash;44. \u003c/li\u003e\n\u003cli\u003eTumial\u0026aacute;n LM, Theodore N. Basilar artery thrombosis after reduction of cervical spondyloptosis: A cautionary report - Case report. J Neurosurg Spine. 2012 May;16(5):492\u0026ndash;6. \u003c/li\u003e\n\u003cli\u003eGasco J, Dilorenzo DJ, Patterson JT. C4-C5 post-traumatic spondyloptosis with in situ fusion: Systematic literature review and case report. Spine (Phila Pa 1976). 2013 May 1;38(10). \u003c/li\u003e\n\u003cli\u003eSaleh S, Swanson KI, Bragg T. Successful surgical repair and recovery in a 2-week-old infant after birth-related cervical fracture dislocation. J Neurosurg Pediatr. 2018 Jan 1;21(1):16\u0026ndash;20. \u003c/li\u003e\n\u003cli\u003eRokaya PK, Khadka NK, Giri PK, Khapung R, Mahaseth N. Burst fracture of c5 with traumatic anterior spondyloptosis of c6 and posterior spondylolisthesis of c4 vertebra: A case report. J Nepal Med Assoc. 2021 Apr 1;59(236):402\u0026ndash;5. \u003c/li\u003e\n\u003cli\u003eOppenlander ME, Hsu FD, Bolton P, Theodore N. Catastrophic neurological complications of emergent endotracheal intubation: Report of 2 cases. J Neurosurg Spine. 2015 May 1;22(5):454\u0026ndash;8. \u003c/li\u003e\n\u003cli\u003eWong K, Chang P, Monasky M, Samuelson R. Traumatic spondyloptosis of the cervical spine: A case report and discussion of worldwide treatment trends. Surg Neurol Int. 2017;8(1). \u003c/li\u003e\n\u003cli\u003eSakti YM, Anzhari S, Kartika A, Irfantian A, Ahmad H, Sakadewa GP, et al. Neglected cervical spondyloptosis of the 5TH \u0026ndash; 6TH cervical spine following cervical manipulation: A case report. Int J Surg Case Rep. 2022 May 1;94:106984. \u003c/li\u003e\n\u003cli\u003eModi JV, Soman SM, Dalal S. Traumatic cervical spondyloptosis of the subaxial cervical spine: A case series with a literature review and a new classification. Asian Spine J. 2016;10(6):1058\u0026ndash;64. \u003c/li\u003e\n\u003cli\u003ePadwal A, Shukla D, Bhat DI, Somanna S, Devi BI. Post-traumatic cervical spondyloptosis: A rare entity with multiple management options. J Clin Neurosci. 2016 Jun 1;28:61\u0026ndash;6. \u003c/li\u003e\n\u003cli\u003eRehman AU, Ahmed A, Zaheer Z, Ahmed B, Lucke-Wold B. International Neurosurgery: The Role for Collaboration. Int J Med Pharm Res. 2023;4(1):15\u0026ndash;24. \u003c/li\u003e\n\u003cli\u003eOkoro EU, Havryliv TS, Smolanka AV SV. \u0026ldquo;Traumatic Cervical Anterior Spondyloptosis: Literature Review and Case Report.\u0026rdquo; SVOA Neurol. 2021;2(3):84\u0026ndash;94. \u003c/li\u003e\n\u003cli\u003eBhojraj SY, Shahane SM. Posttraumatic cervical spondyloptosis at C6-7 with late-onset cord compression: A new clinical entity. Case report. J Neurosurg. 1992;77(5):792\u0026ndash;4. \u003c/li\u003e\n\u003cli\u003eChadha M, Singh AP SA. Spondyloptosis of C6-C7: a rare case report. Chin J Traumatol. 2010;13(6):377\u0026ndash;9. \u003c/li\u003e\n\u003cli\u003eOzdogan C, Gogusgeren MA DM. Posttraumatic cervical spondyloptosis \u0026ldquo;Case Report.\u0026rdquo; Turk J Trauma Emerg Surg. 1999;5:46\u0026ndash;8. \u003c/li\u003e\n\u003cli\u003eNg C, Feldstein E, Spirollari E, Vazquez S, Naftchi A, Graifman G, et al. Management and outcomes of adult traumatic cervical spondyloptosis: A case report and systematic review. Vol. 103, Journal of Clinical Neuroscience. J Clin Neurosci; 2022. p. 34\u0026ndash;40. \u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"hideJournal":true,"highlight":"","institution":"Obafemi Awolowo University Teaching Hospitals Complex","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Cervical spondyloptosis, Neglected, Anterior-approach","lastPublishedDoi":"10.21203/rs.3.rs-3821132/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-3821132/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eObjectives\u003c/h2\u003e \u003cp\u003eTo highlight the incidence, outcome, and peculiar management challenges of cervical spondyloptosis\u003c/p\u003e\u003ch2\u003eMethods\u003c/h2\u003e \u003cp\u003eA retrospective literature review and case study\u003c/p\u003e\u003ch2\u003eResults\u003c/h2\u003e \u003cp\u003eFive cases of subaxial cervical spondyloptosis were identified from the literature and reviewed. Their demographics, mechanisms of trauma, intervention, and outcome were highlighted.\u003c/p\u003e\u003ch2\u003eConclusion\u003c/h2\u003e \u003cp\u003eSubaxial cervical spondyloptosis is relatively uncommonly documented in the literature. The management outcomes are variable, and peculiar management challenges are highlighted in the index case study.\u003c/p\u003e","manuscriptTitle":"Management of Neglected Post-traumatic C4/C5 Spondyloptosis in a Low Resource Setting: a Review of Literature and Case Study.","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-01-02 14:09:59","doi":"10.21203/rs.3.rs-3821132/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"d050c528-7ec2-42d6-9483-3dbffdb8c5c8","owner":[],"postedDate":"January 2nd, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[{"id":27882306,"name":"Neurosurgery"}],"tags":[],"updatedAt":"2024-01-02T14:09:59+00:00","versionOfRecord":[],"versionCreatedAt":"2024-01-02 14:09:59","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-3821132","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-3821132","identity":"rs-3821132","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}