SPINE Volume 34, Number 13, pp 1399 –1401
©2009, Lippincott Williams & Wilkins
Prevalence of Vertebral Pars Defects (Spondylolysis) in
a Population With Osteogenesis Imperfecta
Wiebe C. Verra, Bsc,* Hans J. Pruijs, MD, PhD,* Erik J. Beek, MD, PhD,†
and Rene´ M. Castelein, MD, PhD*
Study Design. We performed a retrospective study of
spinal roentgenograms of a large cohort of patients suf-
fering from osteogenesis imperfecta.
Objective. To determine the prevalence of vertebral
pars defects (spondylolysis) in patients with osteogenesis
imperfecta.
Summary of Background Data. Patients suffering from
osteogenesis imperfecta are known to be prone to patho-
logic fractures. Pathologic fractures due to microtraumas,
repetitive activities, and posture acting together on a con-
genitally weakened pars interarticularis may lead to ver-
tebral pars defects. The prevalence of vertebral pars de-
fects in patients with osteogenesis imperfecta has never
been studied before to our knowledge.
Methods. We performed a retrospective study of pos-
terior-anterior and lateral, standing, roentgenograms of
the spine of patients with osteogenesis imperfecta for the
occurrence of vertebral pars interarticularis defects.
Results. The patients (5.3%) in our series showed ver-
tebral pars defects, this is not significantly different from
the prevalence of vertebral pars defects in the general
population.
Conclusion. 5.3% of the patients with osteogenesis
imperfecta showed vertebral pars defects, this does not
differ significantly from the occurrence in the general pop-
ulation.
Key words: vertebral pars defects, osteogenesis im-
perfecta, spondylolysis, spondylolisthesis, prevalence.
Spine 2009;34:1399 –1401
Osteogenesis imperfecta (OI) is a genetic collagen dis-
order characterized by different degrees of bone fragil-
ity and low bone mass due to a mutation in 1 of the 2
genes that encode the -chains of collagen type 1;
COL1A1 and COL1A2.
1
The disorder affects approx-
imately 1 in 20.000 births.
2
Because of the features of
OI, patients are known to be prone to pathologic frac-
tures.
Defects of the vertebral pars interarticularis, known
as spondylolysis, are generally supposed to be stress
fractures.
3
Although vertebral body deformities, sco-
liosis, kyphosis, and basilar impression are character-
istic spinal manifestations of OI,
4
so far only few cases
of spondylolysis have been described in patients suf-
fering from OI. Besides a few case reports and an in-
cidental description in the literature of patients suffer-
ing from OI with pars defects, to our knowledge a
systematic study concerning the prevalence of verte-
bral pars defects in patients with OI has never been
performed. We performed a retrospective radio-
graphic study in a relatively large group of young pa-
tients with different types of OI to determine the prev-
alence of defects in the pars interarticularis.
Materials and Methods
The Wilhelmina Children’s Hospital of the University Medical
Centre Utrecht, the Netherlands, is a tertiary referral center for
all patients in the Netherlands with osteogenesis imperfecta. As
part of the general work-up of these patients, a standing pos-
terior-anterior and lateral roentgenogram of the spine is taken
to search for vertebral impressions, scoliosis, basilar impres-
sions, and other spinal deformities. All spinal roentgenograms
were examined by an experienced pediatric radiologist (EB)
and judged for the presence of vertebral pars defects, pars elon-
gations, and slipped vertebrae (spondylolisthesis). The severity
of a spondylolisthesis was graded according to Meyerding.
5
Patient’s age at time of the last roentgenogram, type of OI
according to Sillence,
6
and gender were recorded. Since spon-
dylolysis is considered a developmental disorder that does not
occur at a very young age, patients under the age of 6 were
excluded. Statistics were performed with the Statistical Package
of Social Sciences version 12.
Results
Radiograms of 143 patients with OI were available for
analysis. Thirty patients were excluded from the study
because of an age below 6, none of them showed a spon-
dylolysis.
The 113 remaining patients consisted of 61 girls and
52 boys. The average age at the time the evaluated roent-
genograms were made was 12.5 years old (6 –24 years
old).
The occurrence of the different types of OI is summa-
rized in Table 1. Patients with pars defects are summa-
rized in Table 2.
A total of 6 patients with vertebral pars defects and
different types of OI were found in the studied group, this
is 5.3%. In the general population, 4.4% is reported as
prevalence for spondylolysis at the age of 6, with a re-
ported increase to 6.0% at the age of 18.
2
The difference
From the *Department of Orthopedics, Wilhelmina Children’s Hospi-
tal, University Medical Center Utrecht, Utrecht, The Netherlands; and
†Department of Pediatric Radiology, Wilhelmina Children’s Hospital
Utrecht, Utrecht, The Netherlands.
Acknowledgment date: October 21, 2008. Revision date: December 9,
2008. Acceptance date: December 29, 2008.
The manuscript submitted does not contain information about medical
device(s)/drug(s).
No funds were received in support of this work. No benefits in any
form have been or will be received from a commercial party related
directly or indirectly to the subject of this manuscript.
Address correspondence and reprint requests to R. M. Castelein, MD,
PhD, Department of Orthopedics, Wilhelmina Children’s Hospital /
University Medical Center Utrecht, Utrecht, PO Box 85500, 3508 GA
Utrecht, The Netherlands; E-mail: r.m.castelein@umcutrecht.nl
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