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    KISEP Case Reports J Korean Neurosurg Soc 26 146-151, 1997

    An Occult Fracture in a Ankylosing Spondylitis Patient

    Chang Myong Choi, M.D., Ji Ho Yang, M.D., Il Woo Lee, M.D.,Chul Ku Jung, M.D., Joon Ki Kang, M.D.

    Department of Neurosurgery, Taejeon St Marys Hospital,Catholic Universi ty Medical College,Taejeon, Korea

    = Abstract =

    KEY WORDS

    Case Report

    A 61-year-old male fell off a 3-m high roof landing on

    his head and back. The patient was first treated at a localclinic before transfer to our hospital. In the emergency room

    he was stuporous and irritable. Neurological examination

    revealed no motor weakness. Cervical spine X-ray showed

    hyperlordotic curvature and bamboo spine without fracture.

    Brain CT scan showed hemorrhagic hyperdense lesions in

    the right frontal region, the cerebellar vermis and the fourthventricle Fig. 1 . Respiration was controlled with intubation

    and the man was restrained due to uncooperative irritable

    behavior. The patient was treated conservatively with a

    course of bed rest, hypertonic solutions, anticonvulsants,

    and analgesics. He regained the consciousness alert and his

    general condition was improved over next three weeks. He

    then, suddenly became paraplegic during wheel-chair am-

    bulation with assistance by his family members.

    Examination Paraplegia with trace movement to painfulstimuli was observed. No pain or temperature sense below

    the T12 dermatome were evident. Anal and bulobocavernous

    reflexes were also lost. Neuroradiology Review of T-L spine radiographs de-

    monstrated a supspicious transverse linear fracture through

    the disc and juxta-end plate region between the T11 andT12 vertebrae with typical findings of ankylosing spondylitis

    Fig. 2 . An MRI scan revealed posterior compression of

    spinal cord by a bony spicule at the T12 level with signalchange in marrow Fig. 3 .

    Operation The patient underwent small laminectomy

    and posterior spinal fusion with a rod system. There was a

    transverse fracture line extending posteriorly to the laminae

    and spinous process. A bony spicule was impinging upon

    the dura which exhibited bluish discoloration and swelling.A small amount of epidural hematoma was found. There was

    calcification involving apophyseal joints with interspinous

    ligaments and ligamenta flava resulting in ossification of

    the spaces between the dorsal arches. The small bony spicule

    impining upon the dura was removed. A spinal fusion was

    performed with rods on two segments above and three

    segments below the lesion using sublaminar hooks and

    wires. The hook entry site was made by use of an air drill

    Fig. 4 .Postoperative Course The patient was nursed on a conve-

    ntional hospital bed and turned by log rolling. No significant

    improvement was observed in the immediate postoperativecourse. Neurologic symptoms improved gradually and the

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    Fig. 1. CT scan obtained on admission shows hemorrage in the right frontal region, cerebell ar vermis, and the fourth ventricle.

    Fig. 2. Retrospective examination of films on admission showing suspicious transverse linear fracture line through the ossifieddisc space and juxta-end plate region between the T11 and T12 vertebrae of bamboo spine.

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    An Occult Fracture in a Ankylosing Spondylitis Patient

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    Fig. 3. CT and MRI examinations taken after paraplegia. Computerized tomography section through the T12 pedicle showsdisruption of the posterior arch. MRI scan demonstrates marrow change and a bony spicule impinging upon cord withincreased signal intensity in the T12 area.

    Fig. 4. Postoperative radiograph The fractured spine was fused, two levels above and three levels below the lesion, by a rodsystem with sublaminar hooks and wires.

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    Chang Myong Choi Ji Ho Yang Il Woo Lee Chul Ku Jung Joon Ki Kang

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    patient could walk without assistance approximately 3 months

    after the operation. After two years , the independent activities

    of daily living ADL were possible, but the patient com-

    plained of paresthesia and radiculopathy in both legs.

    Discussion

    Ankylosing spondylitis Marie-Strmpel arthritis resultsin development of a rigid spinal column bamboo or poker

    sign , often severly deformed by exaggerated thoracic

    kyphosis and cervical lordosis 25)30) . The ankylosed spine of

    a patient with chronic ankylosing spondylitis is more prone

    to fracture than the normal spine15)

    . Since most afflicted patients remain physically active, the risk of injury causingfracture and spinal contusion is significant 13)14)23)31) . The

    trauma which causes the fracture is often minor. Fourteen

    of 22 fractures resulted from minor falls in the series of

    Thomas et al 27) .

    The pathology of ankylosing spondylitis has been des-

    cribed in detail elsewhere 8). Although diarthrodial extremities

    and spinal joints are frequently involved, the proliferative

    process which results in spinal ankylosis predominently

    affects ligamentous attachments and intervertebral discs.

    Chondroidal metaplasia is followed by calcification andossification involving the apophyseal joints, as well as the

    anulus fibrosus, and the anterior, but not always the posterior,

    longitudinal ligaments 9). Interspinous ligament and ligam-

    entum flavum involvement is not unusual, resulting in

    ossification of the spaces between the doral arches. Apo-

    physeal joint involvement begins with erosive changes that

    progress to cartilage destruction, joint narrowing, and ank-

    ylosis. Serial biopsies have demonstrated that inflammatorysynovial proliferation generates a thickened vascularized

    fibrous layer which is thought to disrupt adjacent cartilage

    and bone 9)11) . P.R. Weintstein et al found 13 of the 20

    fractures and destructive vertebral lesions to be located in

    the intervertebral discs 21) . This finding suggests a selective

    vulnerability of ossified discs to traumatic stress. Disc ossi-fication may be incomplete, with fibrous and cartilaginous

    tissue persisting centrally and posteriorly, creating a weaker

    structure than the adjacent vertebral bodies. Such transdiscalfractures may be more difficult to visualize radiographically

    than fracture lines passing through the bodies or ppedicles.

    Mostoften the posterior elements are fused and fracture

    lines extend posteriorly to the lamina and spinous process31)

    .Ankylosis of the spine results in biochemical alterations

    that predispose the patient to serious spinal injury. Alth-

    ough the degree of precipitating trauma may be small, the

    ensuing damage to the spine and spinal cord may be exten-

    sive 12) . Fractures through the cervical spine in ankylosing

    spondylitis have been well documented in the literature,usually occurring through the ossified disc space or ver-

    tebral body and extending posteriorly through the posterior

    elements 3)19)20)31) . The existence of similar fractures in thethoracic and lumbar spine has not been emphasized as clearly,

    probably because of confusion with inflammatory changes

    as well as the lack of serious neurologic impairment. Lower

    thoracic fractures are also quite unstable, and even minor

    mechanical stresses may cause dangerous dislocation of thefracture margins 6). In the ankylosed spine where compen-satory disc and facet joint movement in response to load is

    prevented, it seems likely that bony deformation and, the-

    refore, stress fractures would tend to occur most frequently

    at the thoracolumbar and lumbosacral junctions 2)7)10)17)29)32) .

    Once a fracture has occurred, the long rigid lever arm

    represented by the ankylosed spine tends to concentrate

    stress and deformation at the fracture site. This may contribute

    subsequently to the formation of pseudoarthrosis. Disc space

    or end plate horizontal fractures with associated posterior

    element involvement are common in long-standing ankylosisspondylitis and reflect the bio-mechanical effect of trauma

    or subclinical stress on a rigid spine 20) .

    An occurrence of minor spinal trauma can lead to problems

    in diagnosis. Fractures in ankylosing spondylitis can be

    overlooked because ligamentous and disc ossification fre-

    quently obscure transverse and oblique fractures on plain

    radiographs 21) . In our case, the delay in diagnosis was due

    to the associated severe head injury which caused the failureof physicians to detect subtle spinal lesion during initial

    evaluation. When a patient is represented to a physician

    who is unfamiliar with this type of problem, the diagnosis

    of spinal fracture may not be considered, or it may be

    dismissed readily when radiographs fail to demonstrate a

    spinal fracture. The difficulty in radiographic visualizationof these fractures is a potentially serious problem 15)27) . A

    delay in correct radiographic diagnosis at emergency room

    can result in subsequent development of spinal cord damage.Delayed onset of neurologic deficit due to instability was

    found to be present in 2 cases out of 13 patients 21). Fractures

    can frequently occur as a result of even minimal trauma

    and may be associated severe neurologic deficits. Fracturesthat occur in ankylosing spondylitis are potentially lethal

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    and associated with severe spinal cord injury. Although the

    majority of fractures occur in the lower cervical spine, they

    may occur at any levels and often result in quadriple-

    gia1)13)17)27)29) . Radiographically, the fractures most commonly

    occur through the intervertebral disc space. However hori-zontal fractures through the vertebral bodies adjacent to the

    vertebral endplate, similar to the Chance or seat belt fra-

    cture, with extension through the posterior elements mayalso occur 21) .

    The ankylosed spine fractures like a long bone, and if

    the fracture is through and through involving the posterior

    elements of the spine, it is considered to be unstable due to

    the loss of ligamentous support3)4)15)27)

    . The loss of normalspinal segmental flexibility and the accompanying osteo-

    porosis predispose the ankylosed spine to injury 22)31) . It is

    essential that this borne in mind during the initial handling

    of the patient and during the period of fracture healing. The

    importance of careful immobilization can not be stressed

    too highly. All patients with known ankylosing spondylitis

    should be warned by their physicians of the inordinate

    susceptibility to spinal fractures. These patients after trauma,

    or even if they note spontaneous back pain associated with

    a jerking or grating sensation, should immobilize themselves

    and immediately seek medical attention, as recommended by Osgood et al. This is most imperative with cervical and

    lower thoracic sensations 18) .

    Radiographic recognition in the patient presenting sudden

    focal pain and tenderness is important for selection of proper

    therapy. If bony irregularity and sclerosis begin to develop,

    a pseudoarthrosis rather than a pyogenic or granulomatous

    infection should be suspected and rigid internal fixation

    and surgical fusion may be required 20)24) . Fractures of thethoracic and lumbar spine in ankylosing spondlyitis generally

    have fewer neurologic complications than cervical fractures

    and heal with moderate immobilization. Conservative treat-

    ment with external support by a brace is associated with a

    high rate of fracture union and a low rate of complications

    and mortality. Surgical intervention may be indicated fortreatment of incomplete or evolving neurologic lesions, and

    in the management of fractures that can not be stabilized

    by nonopera tive means 5) . We think that this case report presents a challenge to physicians for management of brain-

    injury associated ankylosing spondylitis where thorough

    examination of radiographs are mandatory in the initial

    evaluation.

    Conclusion

    Although complications are uncommon from minor spinal

    trauma in patients with ankylosing spondylitis, they should be kept in mind, especially in head injured patients. Thorough

    clinical and roentgenographic examination of the entire ver-

    tebral column is recommended in patients with ankylosingspondylitis who have sustained injury.

    References

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