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LETTERS
Ossification of the posterior longitudinal ligament in
ankylosing spondylitis
To the Editor:
In the February 1983 issue of Arthritis and Rheuma-
tism, Yagan et a1 (1) reported a case of ossification of the
posterior longitudinal ligament (PLL) in a patient with
HLA-B27 positive ankylosing spondylitis (AS). We have
recently seen a similar case, which we report here.
The patient was a 39-year-old man with a 13-year
history of inflammatory low back pain and stiffness, and
decreasing range of motion of the lumbar spine. In the past
few years, his symptoms had extended to the cervical spine.
There was no history of urethritis, diarrhea, psoriasis, periph-
eral arthritis, uveitis, conjunctivitis, or cardiac symptoms.
Physical examination disclosed dorsal kyphosis, for-
ward craning of the cervical spine, and severe restriction of
motion of both the cervical and the lumbar spine, in all
directions. Chest expansion was reduced to 1.5 cm.
Figure 1. Lateral radiographic view of the patient’s cervical spine,
showing squaring of the vertebral bodies, marginal syndes-
mophytes, apophyseal joint sclerosis and fusion, and Posterior
longitudinal ligament (PLL) ossification (white arrows) extending
from C1 to C6. A radiolucent line (black arrowheads) is visible
between the posterior aspect of the vertebral bodies and the ossified
PLL.
Arthritis and Rheumatism, Vol. 31, No. 3 (March 1988)
Laboratory evaluation revealed an erythrocyte sedi-
mentation rate (Westergren) of 39 m d h o u r , a C-reactive
protein level of 3.2 mg/dl (normal <0.5), and a serum IgA
level of 626 mg/dl (normal 50-350). HLA typing was positive
for the B27 antigen.
Radiographic examination of the pelvis showed
ankylosis of both sacroiliac joints, erosions, and irregular
periosteal new bone formation (“whiskering”) at the ischial
tuberosities, together with osteitis of the symphysis pubis.
Radiographs of the lumbar and the dorsal spine showed
squaring of the vertebral bodies and apophyseal joint scle-
rosis and ankylosis. Cervical spine radiographs (Figure 1)
demonstrated squaring of the vertebral bodies, marginal
syndesmophytes, apophyseal joint sclerosis and fusion, and
ossification of the PLL extending from Cl to C6. A radiolu-
cent line was visible between the posterior aspect of the
vertebral bodies and the ossified PLL.
PLL ossification is an uncommon condition with an
unclear etiology. It is most frequently reported in Japanese
people (2,3). In 1978, Resnick et a1 (4) reported finding PLL
ossification in 50% of 74 North American patients who had
diffuse idiopathic skeletal hyperostosis (DISH), and sug-
gested that there was an association between the 2 conditions.
The report by Yagan et al in 1983 (1) described the
first case of PLL ossification in a patient with a clear diagnosis
of B27 positive AS. That patient also had an ossification of the
anterior longitudinal ligament of the cervical spine, similar to
that seen in DISH. Although the patient showed no other
signs of DISH in the thoracic or the lumbar spine, the authors
wondered “whether the ossification of the anterior longitudi-
nal ligament of cervical spine-similar to that seen in diffuse
idiopathic skeletal hyperostosis-contributed to the develop-
ment of PLL ossification in the patient’s neck.” Unlike their
patient, ours showed no signs of DISH in the cervical,
lumbar, or dorsal spine.
This report confirms the hypothesis by Yagan et a1
that PLL ossification and AS may coexist in a patient.
Ignazio Olivieri, MD
Donatella Trippi, MD
Gabriele Gemignani, MD
Giampiero Pasero, MD
University of Pisa
Rossella Grazzini, MD
Santa Chiara Hospital
Pisa, Italy
I. Yagan R, Khan MA, Bellon EM: Spondylitis and posterior
longitudinal ligament ossification in the cervical spine. Arthritis
Rheum 26:226-230, 1983
2. Ono K, Ota H, Tada K, Hamada H, Takaoka K: Ossified
posterior longitudinal ligament: a clinicopathologic study. Spine
2:126-138, 1977
3. Nakanishi T, Mannen T, Toyokura Y, Sakaguchi R, Tsuyama N:
Symptomatic ossification of the posterior longitudinal ligament of
the cervical spine. Neurology 24: 1139-1 143, 1974
4. Resnick D, Guerra J Jr, Robinson CA, Vinct VC: Association of
diffuse idiopathic skeletal hyperostosis (DISH) and calcification
and ossification of the posterior longitudinal ligament. AJR
131:1049-1053. 1978