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. 2017 May 31;7(5):469–481. doi: 10.1177/2192568217700108

Table 2.

Important Studies Identified in the Literature Review Relative to the Numbers of Patients Treated, Treatment Option, Summarized Results, and Delayed Diagnosis of Ankylosing Spondylitis (AS) and Diffuse Idiopathic Skeletal Hyperostosis (DISH) (Oxford Centre for Evidence-Based Medicine; Available at http://www.cebm.net).

First Author, Reference Year Study Location Level of Evidence Study Design Disease Entity Number Of Patients With Fractures Treatment Results
Lukasiewicz22 2016 USA 3 Retrospective cohort study AS 498 patients with cervical spine fractures Surgery and conservative treatment Spinal cord injury in 21% of patients; 13% of patients had fractures in more than one region of the spine; 29% adverse events (urinary tract infections, intubation, pneumonia), 6.6% of patients died during their inpatient stay; high morbidity and mortality of fractures in the ankylosed spine
Ma68 2015 China 3 Retrospective study AS 25 patients with cervical spine fractures Anterior-only stabilization (6); posterior-only stabilization (9); combined posterior-anterior or anterior-posterior stabilization (10); median segments of fixation 4 Surgical treatment advocated, highly unstable cervical spine fractures; looseness or migration of internal fixation was not observe; 14 patients with complications (one death)
Robinson78 2015 Sweden 3 National registry cohort study AS 534 cervical spine fractures (in 990 patients with 1131 fractures) 381 surgery; 153 conservative treatment Clear beneficial effect on survival of AS spinal fractures if treated surgically (long posterior fixation of at least 3 levels above and below the fracture is recommended); spinal cord injury threatened the survival of patients
Xiang72 2015 China 3 Retrospective study AS 11 patients with cervical spine fractures Anterior-posterior stabilization of all fractures No treatment-related injury to the spinal cord, nerve roots, or vessels; no wound infection; primary intention; fracture healing generally required 4-6 months without loosening, no pull-out or collapse of the bone graft, no loosening or breaking of the internal fixators, no pseudarthrosis, or other complications; combination of anterior-posterior approaches seems to be feasible
Schiefer51 2015 USA 3 Retrospective review DISH and AS 36 (39%) cervical spine fractures (of 92 fractures in 81 patients with DISH (42%) or AS (58%) Percutaneous instrumentation (21.7%), posterior instrumented fusion (51.1%) with or without decompression, circumferential decompression and fusion (4.4%), or anterior decompression and fusion (6.6%); remaining patients treated with primary external immobilization Instrumentation over a median of 7 levels; approximately the half of initial conservative treated patients required surgery; neurologic deterioration after presentation in 7 patients (8.6%); 5 of these deteriorated after surgical treatment, 7.6% surgical risk; ASIA grade and patient age predictors of poor outcome at 1 year (P < .001); delayed diagnosis in 20% (19) of the patient cohort (median delay of 8 days)
Prieto-Alhambra6 2014 Denmark 3 Case-control study AS 139 patients with fracture vs 271 controls (age- and gender-matched controls) Not stated Fivefold higher risk of spine fracture for AS patients, peak 2.5 years following diagnosis
Westerveld10 2014 Netherlands 3 Retrospective cohort study AS and DISH 14 patients with AS, 40 patients with DISH; 111 patients in control group Surgical treatment in 8 AS patients, 21 DISH patients, 39 control patients; total of 68 patients with surgery Unstable fractures in AS and DISH; complication rate higher in ASD compared to controls; age and presence of DISH predictors of mortality after a spinal fracture
Gilard31 2014 France 4 Retrospective cases series AS 7 patients with cervical spine fracture or luxation All surgical Mean of 13 days’ delay between trauma and admission; cervical spine fracture in AS with high instability; early surgical treatment due to the risk of secondary neurological deterioration; CT should be the gold standard for exploration of these patients
Mathews43 2013 USA 4 Retrospective cases series AS 6 patients with cervical spine fractures (11 patients) 1 patient posterior instrumentation; 1 patient combined anterior-posterior instrumentation (with discectomy); 1 patient laminectomy with posterior instrumentation; 3 patients conservative treatment No deaths, more nonneurological complications in surgically treated patients (urinary tract infection, pneumonia, cardiac complications)
Hong 63 2013 China 3 Retrospective study AS 8 patients with cervical spine fractures 5 patients anterior instrumentation; 1 patient posterior instrumentation; 1 patient anterior–posterior instrumentation High rate of neurological deficits; surgery should be performed as early as possible
Bransford42 2012 USA 3 Retrospective study DISH 33 patients with cervical spine fractures 16 patients anterior instrumentation, 12 patients posterior instrumentation, 5 patients anterior-posterior instrumentation Average time to diagnosis 2 days; significant experience of neurological injuries of fractures through the disc space (lower ASIA scale), average number of instrumented vertebral bodies 3.6; 5 (15%) patients required reoperation: 2 wound infections (posterior), 1 anterior screw displacement, 1 anterior revision due to unsatisfied reduction, 1 construct failure in a 1-level corpectomy; significant association with lower preoperative neurological status and postoperative complications; mortality of 27% (9 patients; all with neurological deficits; 23 days postinjury)
Backhaus32 2011 Germany 3 Retrospective study AS 51 patients with 51 cervical fractures 31 patients anterior instrumentation, 8 patients posterior instrumentation, 12 patients anterior-posterior instrumentation Early diagnosis of spine fractures with conventional radiographs and CT for adequate treatment; severe neurological complications after dislocation of the fracture; underdiagnosed or underestimated fractures in AS
Caron3 2010 USA 3 Retrospective study AS and DISH 112 patients with a total of 67 cervical spine fractures (28 AS patients and 39 DISH patients with cervical spine fractures) 75 patients treated surgically, 37 patients treated with bracing Majority cervical spine fractures (67 patients; 55%); median time interval from admission to surgical intervention 2 days; most common surgical treatment posterior segmental fixation with at least 3 bilateral points of fixation above and below; higher likelihood of delay in diagnosis and to be treated surgically in AS patients; age and cardiac disease major predictors of mortality; 84% patients with at least 1 complication (pulmonary problem, urinary tract infection)
Sapkas5 2009 Greece 4 Not stated AS 7 patients with cervical spine fractures 4 patients posterior stabilization; 3 anterior-posterior stabilization Spinal fusion in all cases; 100% fusion rate; no intraoperative complications; after surgery, improvement of all patients’ neurological status; loosening of posterior screws without loss of stability in 2 patients; Philadelphia-type cervical collar in all patients postoperatively for 3 to 6 months
Vives86 2008 USA 4 Case report AS 1 patient with a C7 fracture Laminectomy without instrumented fusion followed by a cervicothoracic orthosis postoperatively Progressive weakness over the course of 2-3 hours after admission; spinal cord injury at the C5 level; premedication of clopidogrel (Plavix); C5 and C6 lamina and spinous process fractures with a large epidural hematoma extending from the C3-C7 levels; hematoma evacuation through a laminectomy of C3-C7 without instrumented fusion; use of cervicothoracic orthosis postoperatively; excellent recovery after 18 months without malalignment; presence of syringomyelia on follow-up images
Einsiedel20 2006 Germany 3 Retrospective study AS 37 patients with cervical spine fractures Anterior-only instrumentation (10); single-session anterior-posterior instrumentation (11); 2-session anterior-posterior instrumentation (13); uninstrumented laminectomy (1); no radiologically detected fracture); posterior-only instrumentation (2) Preoperative neurological deficits in 36 patients; 13 patients with delayed diagnosis and more severe neurological deficits and less postoperative improvement compared to prompt diagnosis; 5 patients with early implant failures (all required revision surgery, initially anterior-only instrumentation); 3 deaths due to respiratory distress syndrome (rigid thorax) or cerebral ischemia (rupture of the vertebral arteries); recommendation for anterior decompression and fusion with posterior instrumentation (1- or 2-stage)
Payer71 2006 Switzerland 4 Case series AS 4 patients with cervical fractures Combined anterior-posterior instrumentation Combined anterior-posterior instrumentation of 1 patient after progressive deformity with initial treatment of a halo fixator; combined approach in 1 patient after early redislocation of an anterior monosegmental fixation; 2 patients operated on primarily using the combined approach; good postoperative alignment and stability and no dislocation or deformity during a mean observation period of 11 months with the use of the anterior-posterior approach
Zdichavsky4 2005 Germany 3 Retrospective study AS 32 patients with 19 cervical spine fractures Anterior-only instrumentation (9); posterior-only instrumentation (2); posterior-anterior instrumentation (6); conservative (2) 8 of 17 patients with cervical spine fractures initially treated conservatively were operated on secondarily due to insufficient repositioning (1), instability/fracture dislocation (4), or neurological deterioration (2); banal traumas mostly associated with fractures at the C5/6 and C6/7 levels; anterior-posterior instrumentation better for mobilization and avoiding complications
Cornefjord75 2005 Sweden 3 Retrospective chart review AS 19 patients with cervical fractures Posterior instrumentation (15); anterior-posterior instrumentation (4) 11 patients without neurological deficits preoperatively; 8 patients with neurological deficits preoperatively—improvement of 2 patients postoperatively during their hospital stay, others remained unchanged; no signs of disturbed healing, nonunions or loosening of implants using plain radiographs
Lange66 2005 Germany 4 Case series AS 2 patients with cervical spine fractures 1 patient posterior instrumentation; 1 patient anterior instrumentation; both patients secondary combined posterior-anterior instrumentation due to secondary fracture dislocation Neurological deficit in 1 patient before the initial treatment, with complete recovery after the first treatment postoperatively (anterior-only); second patient without neurological deficit before the dorsal instrumentation—due to fracture dislocation, a second anterior instrumentation was carried out; after combined instrumentation, no dislocation and uneventful healing in both cases
El Masry76 2004 UK 4 Case report AS 1 patient with C7 fracture Anterior-posterior instrumentation (vertebrectomy) No neurological deficit at admission; displaced fracture and running through the right lamina of C7 vertebra; initially hard collar; combined anterior-posterior instrumentation with vertebrectomy; mobilization (2 days after surgery) and Philadelphia collar (for 12 weeks) postoperatively; regular clinical and radiological follow-up; maintenance of anatomical reduction and solid fusion with no shift or breakage of the instrumentation 4-year postoperatively
Moreau73 2003 UK 4 Case series AS 2 patients with cervical spine fractures (one patient with accompanying lumbar fracture) Anterior-posterior instrumentation (1); anterior instrumentation (1) 1 patient with sensory level of T4 and paraplegia of lower extremities (anterior instrumentation); second patient without motor weakness but accompanying lumbar fracture (anterior-posterior instrumentation)
Schröder91 2003 Germany 4 Case series AS 3 patients with cervical fractures Halo treatment 2 pin-track infections and pin protrusion through the skull, with one case of intracerebral hemorrhage and craniotomy; local revision in the second patient
Einsiedel56 2001 Germany 4 Retrospective case series AS 12 patients with cervical spine fractures All surgical 12 patients with cervical fractures, 11 with preoperative neurological deficits; all patients underwent surgery (posterior-anterior instrumentation, anterior stabilization, laminectomy); 5 patients improved after surgery; in 5 patients, the neurological deficits disappeared after surgery; 2 patients died (acute respiratory distress syndrome and cerebral ischemia); 1 patient with revision surgery due to implant dislocation
Taggard77 2000 USA 4 Not stated AS 7 patients with cervical spine fractures Posterior instrumentation 7 patients suffering from AS with cervical fractures; 4 patients were neurologically intact; 3 had quadriparesis; 1 with quadriparesis; all patients treated with posterior stabilization; 4 patients within 2 weeks, 3 patients after failed conservative treatment; no patients with postoperative deterioration; no perioperative complications, 2 patients died of nonsurgical complications during the follow-up
Wu80 1998 Taiwan 4 Case report AS 1 patient with a C6 fracture Laminectomy followed by a halo vest fixation 1 patient with a C6 fracture and a spinal epidural hematoma developed progressive weakness and numbness below C5; laminectomy and hematoma evacuation was performed; neurological function improved after surgery
Olerud41 1996 Sweden 4 Not stated AS 19 patients with cervical fractures 17 patients with anterior or posterior instrumentation, 2 patients with conservative treatment 19 patients with cervical spine fractures, 17 treated surgically with posterior and anterior approaches, 2 patients received halo treatment
Detwiler90 1990 USA 3 Retrospective study AS 11 patients cervical fractures 8 posterior instrumentations with and without decompression 11 patients with cervical spine fractures; all patients initially treated with traction (1 patient with Philadelphia collar); 3 patients died shortly after injury; 8 patients underwent posterior instrumentation; improvement of all neurological deficits in 2 patients; none of the patients deteriorated after surgical treatment
Broom81 1988 USA 4 Case series AS 5 patients with cervical fractures (4 with delayed diagnosis) Not stated 5 patients with cervical spine fractures; all developed delayed neurological complications ranging from 2 to 35 days; the diagnosis of the cervical fracture was delayed in 4 patients

Abbreviations: ASIA, American Spinal Injury Association; CT, computed tomography.