Open Journal of Orthopedics, 2013, 3, 278-282
http://dx.doi.org/10.4236/ojo.2013.36051 Published Online October 2013 (http://www.scirp.org/journal/ojo)
Successful Treatment of Spine Fracture for Diffuse
Idiopathic Skeletal Hyperostosis with Teriparatide—A
Report of Two Cases*
Yasuaki Iida, Hiroshi Takahashi#, Yuichiro Yokoyama, Yasuhiro Inoue, Daisuke Suzuki,
Keiji Hasegawa, Shintaro Tsuge, Wataru Shishikura, Katsunori Fukutake, Ryo Takamatsu,
Kazumasa Nakamura, Masayuki Sekiguchi, Akihito Wada
Department of Orthopaedic Surgery, Toho University School of Medicine, Tokyo, Japan.
Received August 29th, 2013; revised September 28th, 2013; accepted October 6th, 2013
Copyright © 2013 Yasuaki Iida et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
ABSTRACT
We experienced 2 cases of spinal fracture in patients aged 80 years or older with diffuse idiopathic skeletal hyperostosis
(DISH). Since they decided not to undergo surgical treatment, we provided conservative treatment with teriparatide.
There has been no previous study on teriparatide for spine fracture in DISH. This is the first report of the successful use
of teriparatide to treat spine fracture in DISH without surgical intervention. Our patients were treated with teriparatide
for 8 - 9 months after diagnosis of the fracture. Union was obtained in two cases and no adverse events were observed
during treatment. Our report showed that teriparatide could be an alternative to convention al intervention in spin al frac-
ture of DISH.
Keywords: Diffuse Idiopathic Skeletal Hyperostosis; Teriparatide; Spine Fracture
1. Introduction
Patient s with dif fuse i dio pathi c skel etal hype rost osis (DIS H)
may experience problems in that spinal bone fractures
can occur in response to slight external forces. In such
bone fractures, it is difficult to ensure that bone union is
achieved with conservative treatment, as seen in trans-
verse fracture of a long bone.
Since such bone fractur es occur as low-en ergy trauma,
there is a potential that the bone fracture could be over-
looked at the first hospital visit and diagnosed as a dislo-
cation, and that inappropriate treatment would then be
provided.
For bone fractures that occur in patients with ankylos-
ing spinal disorders (ASD), such as ankylosing spondy-
litis (AS) and DISH, surgical treatment is recommended
rather than conservative treatment, based on the results of
analyses of the incidence of complications and prognosis
[1].
However, it has also been reported that the incidence
of postoperative complications and the mortality would
be high because DISH patients tend to be older in many
cases [1,2].
Herein, we report our experience of 2 cases, in which
bone union was achieved by using conservative treatment
with teriparatide, thus, enabling walking with no lumbar
pain. For these cases, we did not select surgical treatment
because the patients were among extreme old age and had
various medical comorbidities.
2. Case 1
An 83-Year-Old Female Patient
The patient had a history of type II diabetes, cerebral in-
farction and cardiac infarction, and she had received in-
sulin therapy for diabetes, and stent placement in the an-
terior descending branch for cardiac infarction.
She visited our hospital for persistent lumbar pain,
which began after she stumbled and fell down in a room.
She had percussion tenderness at the spinous process
in the thoracolumbar spine, and the pain was increased
by body motion.
*Conflict of interest: None.
Financial support: None.
#Corresponding author.
Although plain X-ray fi ndi ngs suggested DISH, no clear
bone fracture was c onfirmed (Figure 1).

Successful Treatment of Spine Fracture for Diffuse Idiopathic Skeletal Hyperostosis with
Teriparatide—A Report of Two Cases 279
Figure 1. Lateral plain X-ray image at the first hospital visit.
Ossification of the anterior longitudinal ligament was con-
firmed in a wide area from the thoracic vertebrae to the
lumbar vertebrae.
MRI showed a fracture line passing from the ossified
area of the anterior longitudinal ligament at Th12/L1 le-
vel through the inferior border of the Th12 vertebral body.
Based on these findings, the patient was diagnosed as
having a bone fracture of DISH caused by a slight exter-
nal force (Figure 2).
The patient was hospitalized for complete rest. Al-
though surgical treat ment was considered for severe pain
in the dorsal position, we began to provide conservative
treatment with teriparatide because the patient and her fa-
mily did not wish her to undergo surgical treatment due
to her extreme old age and the high risk of various medi-
cal comorbidities.
One month after commencement of conservative treat-
ment, she became aware of improvement in the lumbar
pain, and after 2 months, she was able to walk using a
corset and a T-cane.
In CT findings obtained 4 months after development
of the bone fracture, formation of a callus was confirmed,
although local kyphosis was observed at the fracture site,
and the patient could walk for about 5 minutes using a
T-cane. X-ray and CT findings obtained 6 months after
the development of bone fracture showed favorable for-
mation of the callus, and she was aware of no lumbar
pain and was able to climb and descend stairs (Figure 3).
3. Case 2
An 83-Year-Old Male Patient
The patient had a history of type II diabetes, Chronic
Figure 2. Sagittal STIR image of lumbar spine MRI. T1-
weighted image shows a linear low intensity area along the
end plate on the Th12 caudal vertebral body, whereas the
T2-weighted image shows a linear high intensity area. In
the STIR image, a high intensity area was confirmed from
the area along the anterior border of the caudal vertebral
body.
Figure 3. Sagittal CT image. Although local kyphosis was
observed in the fracture site, 6 months after bone fracture
favorable formation of callus was confirmed.
Obstructive Pulmonary Disease (COPD), and cerebral in-
farction. He had received insulin therapy for diabetes and
domiciliary oxygen therapy for COPD.

Successful Treatment of Spine Fracture for Diffuse Idiopathic Skeletal Hyperostosis with
Teriparatide—A Report of Two Cases
280
When he left home, he lost his balance and fell down.
He subsequently developed lumbar pain and difficulty in
walking, and was referred to our hospital.
When he visited our hospital, he had percussion ten-
derness at the spinous process of the lower lumbar spine.
Based on plain X-ray finings, he was diagnosed as hav-
ing DISH, and compressed fracture of the L3, L4 verte-
bral body in the discontinuous ossified site of the anterior
longitudinal ligament was suspected (Figure 4).
MRI showed bone fracture of the L3, L4 vertebral bo-
dy (Figure 5).
He was hospitalized for complete bed rest, and surgi-
cal treatment was considered due to the severe pain in the
dorsal position. After consultation with the patient and
his family regarding therapeutic selection, it was decided
to begin treatment with teriparatide since they did not
wish him to undergo surgical treatment due to the ex-
treme old age and the high risk of medical comorbid ities.
One month after commencement of the treatment with
teriparatide, t he l umbar pain improved, and aft e r 2 months,
the patient was able to walk using a T-cane.
Plain X-ray images taken 6 months after the develop-
ment of the bone fracture showed clear crush of the L3,
L4 vertebral body, and formation of a callus in the ante-
rior longitudinal ligament. Flexion-extension radiograph
Figure 4. Lateral plain X-ray image obtained at the first
hospital visit. The irregular end plate was confirmed on the
inferior border of the L3 vertebral body and the superior
border of the L4 vertebral body.
Figure 5. T1-weighted MR image at the first hospital visit.
The bone fracture was confirmed with a low intensity area
in the T1-weighted image for the L3, L4 vertebral body.
showed no abnormal mobility at the site, and the lumbar
pain was cured, enabling him to be engaged in daily liv-
ing, as he had before development of the bone fracture
(Figure 6).
4. Discussion
DISH is a supposedly non-inflammatory disease in which
the spinal longitudin al ligaments and entheses slowly be-
come ossified. While the clinical entity of DISH was first
described by Resnick et al. [3], its etiology remains un-
known. However, there have been reports of associations
with obesity, type 2 diabetes mellitus and advanced age
[4,5].
According to Westerveld et al., surgical treatment is
recommended for bone fracture in patients with ASD in-
cluding DISH, rather than conservative treatment, based
on the incidence of complicati ons and prognosis [1].
Caron et al. [2] also suggested that the mortality of the
surgically-treated group was 23%, compared to 51%
mortality with no operative treatment, and the mortality
was significantly lower in patients aged 70 years or
older.
Teriparatide is a recombinant bioactive fragment (1-34)
of human parathyroid hormone and is indicated for the
treatment of postmenopausal osteoporosis. It has been
shown to stimulate osteoblasts, enhance bone connecti-
vity, increase endosteal cortical thickness, and improve
bone mineral content [6-10]. The efficacy of teriparatide
on human osteoporosis has already been established, and
many reports have suggested its preventive and analgesic

Successful Treatment of Spine Fracture for Diffuse Idiopathic Skeletal Hyperostosis with
Teriparatide—A Report of Two Cases 281
Figure 6. Plain X-ray findings obtained 6 months after bone
fracture. L3, L4 vertebral body was crushed, but formation
of a callus was confirmed in the anterior longitudinal liga-
ment.
effects for osteoporosis-derived spinal compression frac-
ture [11-13]. In addition, several recent reports have sug-
gested that teriparatide would exhibit efficacy to promote
healing of bone fracture and pseudoarthrosis by aggres-
sively stimulating osteoblasts to promote bone formation
[14-17].
For our 2 cases, we had no choice but to select con-
servative treatment because of the high risk of surgical
treatment and the wishes of the patients and their fami-
lies.
To date, there have been no reports on the use of teri-
paratide in the treatment of spinal fracture associated
with DISH. However, we used the teriparatide to treat
our cases based on the reports of the usefulness for pseu-
doarthrosis at various sites and the promotion of healing
of bone fracture. As a result, both of the patients experi-
enced improved pain at the fracture site at a compara-
tively early phase, and active formation of callus was
confirmed in CT images 4 months after the bone fracture
in one case. Six months after bone fracture, both of the
patients had no lumbar pain and were able to engage in
normal daily activities, as they had before the bone frac-
ture.
For bone fracture in patients with ASD, surgical treat-
ment is recommended, rather than conservative treatment.
However, the incidence of postoperative complications
can be high in patients who undergo surgical treatment.
Teriparatide could be a medicine that will fundamentally
change the concept for conventional treatment methods.
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