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Septic arthritis of the C1C2 lateral facet joint and torticollisPseudo-Grisel's syndrome.

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We present the case of a 76-year-old man who
experienced the sudden development of fever, rightsided neck pain and stiffness, and torticollis. A soft
tissue mass was noted on the right side of his neck, but
his head was tilted to the left. Computed tomography
scans (with reformatted sagittal and coronal images) of
the patient’s cervical spine revealed destructive changes
of the right lateral masses of C1 and C2 and the clivus,
and a well-delineated periodontoid soft tissue mass
(confirmed by magnetic resonance imaging). After the
second episode of right-sided hemiparesis, he underwent
transoral surgical exploration, with anterior decompression and odontoidectomy. Histologic examination of the
surgical material revealed granulation tissue, fibrosis,
and chronic inflammation, consistent with abscess formation with invasion and compression of the spinal cord
and bone. This case suggests that nonreducible rotational head tilt to the side opposite the side of lateral
mass collapse should raise the suspicion of a possible
Pyogenic infections of the spine are usuall’y
attributed to pyogenic vertebral osteomyelitis, which
rarely affects the cervical spine (1-6). Involvement of
From the Department of Radiology, Hendrick Medical
Center, Abilene, Texas, the Department of Medicine, University of
South Alabama, Mobile, and the Department of Medicine, University of Texas Southwestern Medical School, Dallas.
James T. Halla, MD: Abilene, TX: Johnny Bliznak, MD:
Department of Radiology, Hendrick Medical Center; Joe G . Hardin,
MD: Professor of Medicine, University of South Alabama: Sam
Finn, MD: Assistant Professor of Neurosurgery, University of
Texas Southwestern Medical School.
Address reprint requests to James T. Halla, MD, 1927 Pine
Street, Abilene, TX 79601.
Submitted for publication April 5 , 1990; accepted in revised
form July 7, 1990.
Arthritis and Rheumatism, Vol. 34, No. 1 (January 1991)
the occipitoatlantoaxial (OAA) complex and odontoid
process is even more rare (7-12).
Torticollis as a manifestation of cervical spine
infection has been reported, especially in children
(11,13), and it may take the form of Grisel’s syndrome
(lateral subluxation at C 1-C2, but no radiographic
destruction) or it may be secondary to vertebral osteomyelitis (1,11,13-16). Nontuberculous bacterial involvement of the Cl-C2 lateral facet joints with torticollis has not been reported.
We describe a patient who suddenly developed
fever, torticollis, and, subsequently, an erosive arthropathy of the right lateral facet joint of Cl-C2 and the
odontoid process with spinal cord compression. The
patient was found to have a mass of infected granulation tissue. He was treated with transomal anterior
decompression, odontoidectomy, and long-term antibiotics with good results.
Case report. The patient, a 76-year-old man,
had been healthy except for osteoarthritis of the hands
and knees until January 1987, when he presented at
another institution with a 3-day history of a “crick in
my neck.” He had experienced progressive right-sided
neck pain and stiffness, and he had a rotational head
tilt to the left and a right-sided soft tissue neck mass at
the angle of the jaw. He had no history of sore throat,
hoarseness, or dysphagia. At admission, he was febrile
(102°F) and confused, and he reported posterior and
suboccipital neck tenderness and pain. Laboratory
evaluation revealed mild leukocytosis (1 1,000 white
blood cells [WBC]/mm3),and analysis of the cerebrospinal fluid (CSF) revealed 180 WBClpI (100%polymorphonuclear leukocytes), with normal glucose levels and no
growth on routine cultures of CSF samples. Results of
Figure 1. Computed tomography images of the patient’s cervical spine. A, Lateral scout view, showing prevertebral soft tissue thickening from
C1 through C3, decreased space between the occiput and the atlas, and slight vertical atlantoaxial subluxation. B, Coronal view, showing
erosive and destructive changes of the odontoid process and the right atlantooccipital and atlantoaxial joints, with rotatory and lateral
subluxation. The erosive changes of the left atlantooccipital and atlantoaxial joints are quantitatively less, but are not well demonstrated on this
image. Inset, Coronal view of the atlantoaxial joint.
blood cultures were unremarkable. A routine radiograph of the cervical spine was interpreted as “normal,” and results of a computed tomography (CT)
scan of the head and cervical spine (without reformatted images) were considered unremarkable. Magnetic
resonance imaging (MRI) was not done.
Cellulitis and/or acute parotitis were considered, and treatment with cefuroxime, intravenously,
was begun, with apparent clinical improvement; oral
antibiotics were continued for 2 weeks thereafter.
Although he became afebrile, the neck pain
increased, lateral rotation decreased, and head tilt to
the left worsened. When fever recurred 5 weeks later,
he was admitted to another hospital, where a technetium diphosphonate bone scan revealed increased uptake in the area of the craniocervical junction; however, results of a gallium scan were negative. After
otolaryngologic consultation, subaxial neck exploration was performed to rule out a malignant process;
this yielded negative findings. Cultures of surgical
specimens were negative.
Because of the negative findings at surgery, a
rheumatologic consultation was obtained. CT scans of
C 1-C2, with axial, reformatted sagittal and coronal
images, and a scout view, showed erosive changes of
the Cl-C2 lateral facet joints, more so on the right
side, with rotatory and lateral subluxation and a prevertebral soft tissue mass (Figures 1A and B), although
the patient had improved clinically. Three weeks later,
however, the erythrocyte sedimentation rate (Westergren) was elevated. CT scans taken at that time
revealed further destructive changes of the right lateral
masses of C1 and C2 and the clivus, and a welldelineated periodontoid soft tissue mass was noted
(Figures 2A and B). MRI confirmed these findings.
The patient suddenly developed right-sided
hemiparesis and truncal paresthesias, and he was
referred to the neurosurgical unit of Parkland Hospital, Southwestern Medical School (Dallas, TX). A
cervical myelogram (with metrizamide contrast medium) and CT scan revealed subtle extradural compression of neural elements at the foramen magnum
and a periodontoid soft tissue mass, but a diagnosis
was still not apparent. Subsequently, a mass was
Figure 2. Computed tomography images taken 3 weeks after those shown in Figure 1. A, Sagittal view, showing a large periodontoid soft tissue
mass, with destructive changes of the odontoid process and anterior arch of the atlas. Inset, Sagittal view of the atlantoaxialjoint. B, Coronal
view, showing a rotational deformity to the left. Progressive destructive changes of the odontoid process, destructive changes at both the
atlantooccipital and atlantoaxialjoints, with greater involvement of the right side than the left, and right lateral mass collapse are evident. Inset,
Coronal view of the atlantoaxial joint.
palpated transorally, and transoral drainage of the
retropharyngeal area yielded 4 cc of purulent material,
cultures of which grew Staphylococcus a w e u s . Blood
cultures were repeatedly negative. The patient was
treated with intravenous nafcillin and fitted with a
Philadelphia collar, and his condition improved. He
experienced less neck pain and there was stabilization
of his neurologic status. However, he again developed
right-sided hemiparesis, and transoral surgical exploration with anterior decompression and odontoidectomy were performed. No purulent material was obtained, but there was erosion of the odontoid process
and bony destruction of the right lateral masses of C1
and C2 and the clivus. Histologic examination of the
surgical material demonstrated granulation tissue, fibrosis, and chronic inflammation, consistent with abscess formation invading and compressing the spinal
cord and bone. All cultures at this time were negative.
Antibiotic therapy was changed to cloxacillin, and the
patient was placed in halo traction.
The patient’s condition steadily improved. After 6 months, the halo traction was removed, and the
cloxacillin was discontinued. CT scans taken at 1 year
and 2 years of followup have shown stabilization of the
changes (Figure 3). The patient remains well, and
there are no signs of myelopathy or neck pain.
Discussion. The OAA complex consists of a set
of synovial articulations at the atlantooccipital, atlantoaxial, and atlantoodontoid joints. The joints work as
a unit, but rotation occurs predominantly at the atlantoaxial joint, and lateral flexion occurs at the atlantoocipital joints. Involvement of this area, which has
received considerable attention recently, is manifested
by a characteristic clinical profile in both noninfectious
inflammatory and noninflammatory arthropathies (162 1). This clinical profile includes nonreducible rotational head tilt (NRRHT), which is the characteristic
physical finding in patients with lateral mass collapse,
and the head tilt is always toward the side of the lateral
mass collapse. The case reported here suggests that
NRRHT to the contralateral side in a patient with
destructive arthropathy should raise the possibility of
Pyogenic spondylitis is defined by Resnick as
spinal osteomyelitis and disc space infection together
(16). Pyogenic spondylitis of the OAA complex has
rarely been reported, and such involvement may be
tuberculous (22-24) or pyogenic (7-12). Tuberculosis
Figure 3. Computed tomography image taken 2 years after those
shown in Figures I and 2. There are destructive changes and fusion
of the atlantooccipital joint, with some reconstitution of the distorted C2 lateral masses. There is marked irregularity of the CI-C2
articulations, with surgical absence of most of the odontoid process.
These changes had remained stable for 2 years. Inset, Coronal view
of the atlantoaxial joint.
involving the atlantoaxial region is very rare but may
be associated with torticollis; nontuberculous pyogenic spondylitis of the OAA complex is even more
rare. Six adult patients with pyogenic spondylitis have
been described (7-10,12), and all but 1 of them had a
predisposing illness ( 3 had diabetes, 1 used intravenous drugs, and 1 had a history of peritonsillar abscess). Neck pain was of sudden onset in 3 , gradual
onset in 2, and prolonged in 1. Three patients were
febrile, and none had clinical signs or symptoms of
spinal cord impingement. Radiographic findings revealed involvement of the odontoid process alone in 2
patients, the body of C2 and the odontoid process in 2
patients, and the CI-C2 lateral facet joint, with lateral
subluxation, and involvement of the odontoid process
in 2 patients. No patient demonstrated torticollis on
physical examination. Four patients had evidence of a
retropharyngeal or peritonsillar abscess.
Torticollis is a rotational deformity of the cervical spine resulting in malpositioning of the head and
neck, and it results from a variety of causes (25).
Torticollis secondary to nontuberculous pyogenic cervical osteomyelitis has not been associated with destructive changes in the OAA complex, although at-
lantoaxial dislocation has been described in children
(11,13). Grisel’s syndrome occurs in children and is
the concurrence of lateral atlantoaxial subluxation and
torticollis, caused by localized infection of adjacent
neck tissue that spreads via the lymphatic system or,
more likely, the pharyngovertebral veins (1 1,13-16).
While no destructive changes are noted radiographically in the OAA complex, hyperemic osteoporosis
and ligamentous laxity are standard findings. Our
patient had fixed torticollis, or NRRHT, with destructive changes at the OAA complex. Unlike previous
descriptions in which NRRHT was toward the side of
the lateral mass collapse, our patient had NRRHT
toward the side opposite the lateral mass collapse.
The diagnosis of septic arthritis and osteomyelitis of the OAA complex was complicated by the
abnormal position of the patient’s head. As we have
reported elsewhere, CT with reformatted images
proved a useful diagnostic tool for this region (20). The
first C T scan performed on this patient did not include
reformatted images and was compromised by the
patient’s abnormal, and painful, head position.
The best procedure for early diagnosis of infectious or noninfectious disease of the Cl-C2 lateral
facet joint is not yet clear. It is apparent that failure to
recognize involvement of the OAA complex may delay
diagnosis and have potentially serious consequences.
Even though our experience with MRI is limited and
we have been particularly pleased with the use of CT
imaging of the OAA complex, MRI may be the preferred imaging method because of the excellent soft
tissue contrast that is achievable (17).
I . Forsythe M, Rothman RH: New concepts in the diagnosis and treatment of infections of the cervical spine.
Orthop Clin North Am 9:1039-1051, 1978
2 . Stauffer RN: Pyogenic vertebral osteomyelitis. Orthop
Clin North Am 6:1015-1027, 1975
3. Frederickson B , Hansen Y, Olans R: Management and
outcome of pyogenic vertebral osteomyelitis. Clin Orthop 131:16&167, 1978
4. Slagel SA, Skiendyielewski JJ, McMurry FG: Osteomyelitis of the cervical spine: reversible quadraplegia resulting from Philadelphia collar placement. Ann Emerg
Med 144:912-915, 1985
5. Musher DM, Thorsteinsson SB, Minuth JN, Luchi RJ:
Vertebral osteomyelitis, still a diagnostic pitfall. Arch
Intern Med 136:105-110, 1976
6. Lowry LD: Osteomyelitis of the cervical vertebrae.
Trans Pa Acad Ophthalmol Otolaryngol 30: 173-177,
7. Venger BH, Musher DM, Brown EW, Baskin DS:
Isolated C2 osteomyelitis of hematogenous origin: case
report and literature review. Neurosurgery 18:461-464,
8. Frank TJF: Osteomyelitis of the odontoid process of the
axis. Med J Aust 1:198-201, 1944
9. Rimolovski AB, Aronson SM: Abscess of medulla oblongota associated with osteomyelitis of odontoid process. J Neurosurg 29:97-101, 1968
10. Ahlback S, Collert S: Destruction of the odontoid process due to atlantoaxial pyogenic spondylitis. Acta Radiol 10:39&400, 1970
11. Sullivan AW: Subluxation of the atlantoaxial joint: sequel to inflammatory processes of the neck. J Pediatr
35:451-464, 1949
12. Leach RE, Goldstein HH, Younger D: Osteomyelitis of
the odontoid process. J Bone Joint Surg 49A:369-371,
13. Visudhiphan P, Chiemchanya S, Somburanasin R, Dheandhanoo D: Torticollis as the presenting sign in cervical
spine infection and tumor. Clin Pediatr 21:71-76, 1982
14. Parke WW, Rothman RH, Brown MD: The pharyngovertebral veins: an anatomical rationale for Grisel’s
syndrome. J Bone Joint Surg 66A:568-574, 1984
15. Odelberg-Johnson G: A case of cervical spondylarthritis
after tonsillectomy. Acta Orthop Scand 2:302-317, 1931
16. Resnick D: Osteomyelitis, septic arthritis and soft tissue
infection: the axial skeleton, Diagnosis of Bone and
Joint Disorders. Second edition. Edited by D Resnick, G
Niwayama. Philadelphia, WB Saunders, 1988
17. Halla JT, Hardin JG, Vitek J, Alarcon GS: Involvement
of the cervical spine in rheumatoid arthritis. Arthritis
Rheum 32:652-659, 1989
18. Halla JT. Fallahi S, Hardin JG: Nonreducible rotational
head tilt and lateral mass collapse: a prospective study
of frequency, radiographic findings, and clinical features
in patients with rheumatoid arthritis. Arthritis Rheum
25:1316-1324, 1982
19. Halla JT, Fallahi S, Hardin JG: Nonreducible rotational
head tilt and atlantoaxial lateral mass collapse: clinical
and roentgenographic features in patients with juvenile
rheumatoid arthritis and ankylosing spondylitis. Arch
Intern Med 143:471-474, 1983
20. Halla JT, Bliznak J, Hardin JG: Involvement of the
craniocervical junction in Reiter’s syndrome. J Rheumato1 15:1722-1725, 1988
21. Halla JT, Hardin JG Jr: Atlantoaxial (Cl-C2) facet joint
osteoarthritis: a distinctive clinical syndrome. Arthritis
Rheum 30577-582, 1987
22. Dowd CF, Sartoris DT, Haghighi P, Resnick D: Case
report 344. Skeletal Radio1 15:65-68, 1986
23. Resnick D: Osteomyelitis, septic arthritis, and soft tissue infection: the organisms, Diagnosis of Bone and
Joint Disorders. Second edition. Edited by D Resnick, G
Niwayama. Philadelphia, WB Saunders, 1988
24. Fang D, Leong JLY, Fang HSY: Tuberculosis of the
upper cervical spine. J Bone Joint Surg 65B:47-50, 1983
25. Bland J: Differential diagnosis and specific treatment,
Disorders of the Cervical Spine. First edition. Edited by
J Bland. Philadelphia, WB Saunders, 1988
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septic, facer, syndrome, grisea, lateral, joint, arthritis, torticollispseudo, c1c2
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