Lenke, Lawrence G.

Riew, K. Daniel

Rudzki, Jonas R.

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Washington University School of MedicineDigital Commons@BeckerOpen Access Publications11-1-2004Pseudarthrosis of a thirty-nine-year-old densfracture causing myelopathyJonas R. RudzkiWashington University School of Medicine in St. LouisLawrence G. LenkeWashington University School of Medicine in St. LouisK. Daniel RiewWashington University School of Medicine in St. LouisFollow this and additional works at: http://digitalcommons.wustl.edu/open_access_pubsPart of the Medicine and Health Sciences CommonsThis Open Access Publication is brought to you for free and open access by Digital Commons@Becker. It has been accepted for inclusion in OpenAccess Publications by an authorized administrator of Digital Commons@Becker. For more information, please contact engeszer@wustl.edu.Recommended CitationRudzki, Jonas R.; Lenke, Lawrence G.; and Riew, K. Daniel, ,"Pseudarthrosis of a thirty-nine-year-old dens fracture causingmyelopathy." The Journal of Bone and Joint Surgery.86,11. 2509-2513. (2004).http://digitalcommons.wustl.edu/open_access_pubs/1070COPYRIGHT © 2004 BY THE JOURNAL OF BONE AND JOINT SURGERY, INCORPORATEDPseudarthrosis of a Thirty-nine-Year-Old Dens Fracture Causing MyelopathyA CASE REPORTBY JONAS R. RUDZKI, MD, MS, LAWRENCE G. LENKE, MD, KATHY BLANKE, RN, AND K. DANIEL RIEW, MD Investigation performed at the Washington School of Medicine, Saint Louis, Missourireatment of fractures of the odontoid process of theaxis (the dens) has the potential to stabilize a criticalinjury or result in a high degree of morbidity or evendeath. We present the case of a patient in whom myelopathydeveloped more than thirty-nine years after he sustained adens fracture while playing football. This case is unique be-cause of the prolonged asymptomatic interval between the in-jury and the development of the myelopathy, during whichtime the patient worked as a manual laborer. The late onset ofprogressive myelopathy after a dens fracture is considered un-common1,2 and, to our knowledge, this case represents the sec-ond longest delay between a dens fracture and the onset ofmyelopathy in the English-language literature. The myelopa-thy resolved after surgical stabilization, and the four-year clin-ical and radiographic follow-up data are presented. Ourpatient was notified that data concerning this case would besubmitted for publication.Case Reportfifty-five-year-old man who performed manual labor work-ing as a machine operator presented to an orthopaedic sur-geon with a chief symptom of difficulty using his upper andlower limbs. He reported the insidious onset of increasing neckpain at the base of the skull over the preceding several years,which had been associated with progressively worsening bilat-eral upper and lower-extremity weakness for the previous threemonths. The patient had been unable to work or participate inhis normal activities of daily living for approximately twomonths because of the arm weakness. The musculoskeletal his-tory revealed that he had sustained a cervical spine fracture atthe age of sixteen years while playing football, which was treatedwith a neck brace for approximately three months.On physical examination, the patient appeared to be awell-developed, well-nourished man with a range of motionof the neck of 45° of rotation bilaterally as well as the ability toperform chin-to-chest flexion and vertical extension to neu-tral. Marked weakness was identified in the upper and lowerextremities bilaterally, with 3+ to 4− (of 5) global strength ofthe upper extremities and 4+ to 5− global strength of thelower extremities. The reflexes were symmetrical in the upperand lower extremities, and the patient had continuous clonusin the right ankle with three beats in the left ankle. He haddecreased light-touch sensation in the ulnar distribution ofthe left upper extremity. The results of Babinski testing wereequivocal bilaterally. Proprioception was within normal limitsin the fingers and toes bilaterally, and the gait was normal. TheHoffmann sign was negative, and he had no evidence of diffi-culty performing rapid alternating movements. Anteroposterior, lateral, and flexion-extension radio-graphs of the cervical spine demonstrated a chronic Andersonand D’Alonzo3 type-II dens fracture with marked subluxationof 2 cm (Figs. 1-A and 1-B). Forward flexion made the sublux-ation slightly worse, whereas extension reduced it by approxi-mately 1 cm. Magnetic resonance imaging of the cervical spinerevealed a reduced space for the spinal cord, with several viewsrevealing a tight constriction of the spinal canal at the C1-C2cervical vertebral levels as well as mild C3-C4 stenosis (Figs. 2-A,2-B, and 2-C). The diameter of the space available for the spi-nal cord measured 5 × 12 mm at the level of the most severestenosis, and a signal change indicative of chronic spinal cordirritation was identified at this level. The patient was diagnosed as having an ununited type-II dens fracture with anterior subluxation and spinal cord im-pingement resulting in progressive myelopathy. He was takento the operating room for placement of a halo vest and subse-quent closed reduction under cervical traction while he wasawake. The halo ring was placed and, under fluoroscopic guid-ance, gentle traction was added in 5-lb (2.3-kg) increments toa total of 25 lb (11.3 kg). By keeping the neck in extension, itwas possible to partially reduce the subluxation. The partialreduction was confirmed radiographically, and the halo ringwas secured to the vest. For definitive treatment, the patient underwent open re-duction of the C2 vertebral fracture-subluxation through aposterior approach two days later. A posterior arthrodesis ofC1 and C2 was accomplished with use of autogenous iliaccrest bone graft and was secured with atlantoaxial cable wir-ing. Open reduction was accomplished by posterior transla-tion of the C1 arch by means of the sublaminar cables, and a4.0-diameter fully threaded transarticular screw was placedTA TH E JO U R NA L OF BONE & JOINT SURGER Y ·  JBJS .ORGVOLUME 86-A ·  NU M B ER 11 ·  NOVEM B ER 2004PSEUDAR THROSIS  OF A TH I R T Y-NINE-YEAR-OLD DENS FR A C TU RE CA U S I N G MYE L OPATHYthrough the right C2 vertebral pars interarticularis across theC1 and C2 vertebral facets for fixation. The patient toleratedthe procedure well and had an uneventful postoperativecourse. Intravenous dexamethasone was administered in theperioperative period. As a result of the degree of force re-quired to reduce the fracture and the relatively poor bone den-sity, it was thought that the patient would benefit from the useof the halo vest for an additional ten weeks. The upper andlower-extremity strength returned to normal within threemonths postoperatively and the clonus resolved as well, allow-ing him to return to work as a laborer.Four years after the spine operation, the patient re-turned for follow-up and reported new-onset left-sided neckpain. He stated that he had no weakness or paresthesias in theupper or lower extremities. On physical examination, he hadapproximately 40° of neck rotation bilaterally, was able toperform chin-to-chest flexion, and had vertical extension ofapproximately 20° past neutral. The neurologic examinationrevealed 5 of 5 upper and lower-extremity strength bilaterallywith a normal gait. Radiographs revealed a solid C1-C2 fusionwith no evidence of abnormal alignment or instability (Figs.3-A and 3-B). A cervical magnetic resonance imaging scanmade at this time revealed adequate space for the spinal cordand cervical roots, but there was mild multilevel spondylosisand signal change in the cervical spinal cord consistent withmyelomalacia. The patient was referred to a physical therapist,and the pain resolved without additional intervention.Discussionens fractures account for approximately 7% to 13% of allcervical spine fractures4,5. An estimated 25% to 40% of in-dividuals who sustain this injury do not survive6,7. It has beenestimated that only 65% of those who do survive the initial in-jury seek medical advice, primarily because of neck pain8, andeven with prompt diagnosis of the injury only 18% to 25% arefound to have immediate neurological disability2. The naturalhistory of untreated dens fractures has not been definitively elu-cidated. The literature contains reports of delayed-onset myel-opathy as long as forty-five years after the fracture1,2. The Anderson and D’Alonzo classification system, whichis based on the location of the fracture, has been valuable inrelation to making a prognosis and planning management ofthe injury3. The most common type of dens fracture is typeII2,9. The mechanism of injury resulting in dens fractures hasDFig. 1-ANeutral (Fig. 1-A) and flexion (Fig. 1-B) plain lateral radiographs made at presentation of the myelopathy. The maximal atlantodental interval on the neutral radiograph measures 4 mm, and the space available for the cord measures 6 mm. Fig. 1-B TH E JO U R NA L OF BONE & JOINT SURGER Y ·  JBJS .ORGVOLUME 86-A ·  NU M B ER 11 ·  NOVEM B ER 2004PSEUDAR THROSIS  OF A TH I R T Y-NINE-YEAR-OLD DENS FR A C TU RE CA U S I N G MYE L OPATHYbeen studied extensively10-14. In series ranging in size from fif-teen to 227 dens fractures, rates of nonunion have rangedfrom 5% (in a series of sixty-three fractures reported byAmyes and Anderson15) to 64% (in a series of twenty-two frac-tures described by Schatzker et al.11). Nonunion rates betweenthese extremes have included 28% in a series of thirty-fivefractures reported by Anderson and D’Alonzo3 and 44% in aseries of twenty-six fractures described by Nachemson8. Thenonunion rate following nonoperative management of type-IIfractures was 20% in a series of forty-six fractures reported bySeybold and Bayley16, 32% in a series of ninety-six patients re-port by Clark and White10, 49% in a series of thirty-three frac-Fig. 2-BFig. 2-CFig. 2-AFigs. 2-A, 2-B, and 2-C T2-weighted magnetic resonance images demonstrating a displaced type-II dens fracture with canal compromise and bright spinal cord signal change consis-tent with edema. The anteroposterior space available for the cord measures 5 mm.  TH E JO U R NA L OF BONE & JOINT SURGER Y ·  JBJS .ORGVOLUME 86-A ·  NU M B ER 11 ·  NOVEM B ER 2004PSEUDAR THROSIS  OF A TH I R T Y-NINE-YEAR-OLD DENS FR A C TU RE CA U S I N G MYE L OPATHYFig. 3-ALateral extension (Fig. 3-A) and flexion (Fig. 3-B) plain radiographs made at the time of the four-year follow-up, demonstrating a solid C1-C2 fusion.Fig. 3-Btures reported by van Holsbeeck et al.17, and 100% in a seriesof fifteen patients reported by Maiman and Larson18. In a case-control study of thirty-three patients with a type-II dens frac-ture treated with halo immobilization, patients older than fiftyyears of age were found to be at a significantly increased riskfor failure of treatment with halo immobilization (p =0.002)19. A retrospective review of 229 C2 fractures by Hadleyet al. revealed that the rate of nonunion of acute type-II frac-tures is highest in patients older than sixty years of age with ≥6mm of displacement4. The nonunion rate for the eighty-seventype-II dens fractures in that study was 28%. Several factorslimit the ability to extrapolate the actual nonunion rate ofdens fractures from the literature; these include a lack of uni-formity with regard to sample sizes, inclusion and exclusioncriteria, radiographic methods, criteria for identification ofnonunion, and methods of external immobilization. The case of our patient is unique because of the long in-terval between the injury and presentation of the myelopathy,the degree of progressive myelopathy developing almost fortyyears after the initial injury, the degree of displacement withoutadditional trauma, and the patient’s complete recovery. Withcareful diagnostic evaluation and thoughtful operative planningfor stabilization of the injury, the patient had full resolution ofthe myelopathy and was able to return to normal activities ofdaily living without restriction. Late-onset myelopathy hasrarely been described in the literature1,2. The asymptomaticinterval of approximately forty years until the onset of themyelopathy secondary to the pseudarthrosis in our patient un-derscores the risks of nonoperative management as well as thepotential for late neurologic compromise. Jonas R. Rudzki, MD, MSLawrence G. Lenke, MDKathy Blanke, RNK. Daniel Riew, MDDepartment of Orthopaedic Surgery, Washington University School of Medicine, One Barnes-Jewish Hospital Plaza, Suite 11300, West Pavilion, St. Louis, MO 63110. E-mail address for L.G. Lenke: lenkel@msnotes.wustl.eduThe authors did not receive grants or outside funding in support of their research or preparation of this manuscript. They did not receive pay-ments or other benefits or a commitment or agreement to provide such benefits from a commercial entity. No commercial entity paid or directed, or agreed to pay or direct, any benefits to any research fund, foundation, educational institution, or other charitable or nonprofit organization with which the authors are affiliated or associated. TH E JO U R NA L OF BONE & JOINT SURGER Y ·  JBJS .ORGVOLUME 86-A ·  NU M B ER 11 ·  NOVEM B ER 2004PSEUDAR THROSIS  OF A TH I R T Y-NINE-YEAR-OLD DENS FR A C TU RE CA U S I N G MYE L OPATHYReferences1. Paradis GR, Janes JM. Posttraumatic atlantoaxial instability: the fate of the odontoid process fracture in 46 cases. J Trauma. 1973;13:359-67. 2. Crockard HA, Heilman AE, Stevens JM. Progressive myelopathy secondary to odontoid fractures: clinical, radiological, and surgical features. J Neuro-surg. 1993;78:579-86. 3. Anderson LD, D’Alonzo RT. Fractures of the odontoid process of the axis. J Bone Joint Surg Am. 1974;56:1663-74. 4. Hadley MN, Dickman CA, Browner CM, Sonntag VK. Acute axis-fractures: a review of 229 cases. J Neurosurg. 1989;71:642-7. 5. Puttlitz CM, Goel VK, Clark CR, Traynelis VC. Pathomechanisms of failures of the odontoid. Spine. 2000;25:2868-76. 6. Bucholz RW, Burkhead WZ, Graham W, Petty C. Occult cervical spine inju-ries in fatal traffic accidents. J Trauma. 1979;19:768-71. 7. Huelke DF, O’Day J, Mendelsohn RA. Cervical injuries suffered in automobile crashes. J Neurosurg. 1981;54:316-22. 8. Nachemson A. Fracture of the odontoid process of the axis: a clinical study based on 26 cases. Acta Orthop Scand. 1960;29:185-217. 9. Benzel EC, Hart BL, Ball PA, Baldwin NG, Orrison WW, Espinosa M. Frac-tures of the C-2 vertebral body. J Neurosurg. 1994;81:206-12. 10. Clark CR, White AA 3rd. Fractures of the dens. A multicenter study. J Bone Joint Surg Am. 1985;67:1340-8. 11. Schatzker J, Rorabeck CH, Waddell JP. Fractures of the dens (odontoid pro-cess). An analysis of thirty-seven cases. J Bone Joint Surg Br. 1971;53:392-405. 12. Alker GJ Jr, Oh YS, Leslie EV. High cervical spine and craniocervical junction injuries in fatal traffic accidents: a radiological study. Orthop Clin North Am. 1978;9:1003-10. 13. Althoff B. Fracture of the odontoid process. An experimental and clinical study. Acta Orthop Scand Suppl. 1979;177:1-95.14. Mouradian WH, Fietti VG Jr, Cochran GV, Fielding JW, Young J. Fractures of the odontoid: a laboratory and clinical study of mechanisms. Orthop Clin North Am. 1978;8:985-1001. 15. Amyes EW, Anderson FM. Fracture of the odontoid process. Report of sixty-three cases. Arch Surg. 1956;72:377-93. 16. Seybold EA, Bayley JC. Functional outcome of surgically and conservatively managed dens fractures. Spine. 1998;23:1837-46. 17. van Holsbeeck E, Stoffelen D, Fabry G. Fractures of the odontoid process. Conservative and operative treatment. Prognostic factors. Acta Orthop Belg. 1993;59:17-21. 18. Maiman DJ, Larson SJ. Management of odontoid fractures. Neurosurgery. 1982;11:471-6. 19. Lennarson PJ, Mostafavi H, Traynelis VC, Walters BC. Management of type II dens fractures: a case-control study. Spine. 2000;25:1234-7. 

Pseudarthrosis of a thirty-nine-year-old dens fracture causing myelopathy

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Pseudarthrosis of a thirty-nine-year-old dens fracture causing myelopathy

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