Open Journal of Orthopedics, 2013, 3, 217-219
http://dx.doi.org/10.4236/ojo.2013.35040 Published Online September 2013 (http://www.scirp.org/journal/ojo)
217
Bone Pathology: Osteopoikilosis
Tolga Tolunay1, Arslan Kaan Arslan1, Izzet Bingol2*, Vedat Bicici2, Mehmet Eren1,
Cheikh Moustapha Mohamed Lemine3, Kasim Kilicarslan2
1Department of Orthopaedics and Traumatology, Yenimahalle Public Hospital, Ankara, Turkey; 2Department of Orthopaedics and
Traumatology, Ankara Ataturk Training and Research Hospital, Yildirim Beyazit University, Ankara, Turkey; 3Department of Or-
thopaedics and Traumatology, Centre Hospitalier National, Nouakchott, Mauritania.
Email: tolgatolunay@hotmail.com, arslankagan@gmail.com, *dr.izzetbingol@hotmail.com, dr_vedatbicici@yahoo.com,
drmehmeteren@hotmail.com, cerebih@yahoo.fr, kilicarslan44@hotmail.com
Received July 15th, 2013; revised August 14th, 2013; accepted August 31st, 2013
Copyright © 2013 Tolga Tolunay 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
Osteopoikilosis is a rare benign sclerotic bone dysplasia. The disease which usually develops asymptomatically is coin-
cidentally diagnosed with direct radiographs. Our subject, a 52-year-old male was admitted to our policlinic two days
ago due to turning of his left ankle causing pain and swelling on his left ankle. We asked for bilateral direct radiograph
of left ankle. The left ankle radiograph indicates multiple, small oval/round hyperdense sclerotic regions. As a result of
available clinical finding and radiological viewing, the patient was diagnosed with osteopoikilosis.
Keywords: Osteopoikilosis; Bone Pathology; Hyperdense Sclerotic Regions
1. Introduction
Osteopoikilosis is a rare benign sclerotic bone dysplasia.
Autosomal dominant inheritance is observed, and the
cause of the pathology is unknown [1]. The disease
which usually develops asymptomatically is coinciden-
tally diagnosed with direct radiographs [2]. In this case
presentation, we discuss a subject who was admitted to
our policlinic due to turning of his left ankle causing pain
and swelling on the left ankle, and who was then diag-
nosed with osteopoikilosis by means of direct radio-
graphs.
2. Case Presentation
The patient, a 52-year-old male was admitted to our poli-
clinic two days ago due to turning of his left ankle caus-
ing pain and swelling on his left ankle. His family had no
history suggestive of osteopoikilozis. He had a history of
hyperthyroidism and thyroid medication use. Orthopae-
dic examination indicated that he had swell, oedema and
tenderness on left ankle medial malleolus level. Anterior
tensile test was negative and the medial malleolus level
did not have any gap. Inversion stress test (Varus stress
test) and Peroneal tendon instability test were negative.
Bilateral direct radiograph was asked for the patient. The
left ankle radiograph did not show any finding which
corresponds to fracture but the bone structures contained
multiple, small oval-round hyperdense sclerotic regions.
In order to find out the distribution of lesions, we asked
for radiographs of bilateral ankle, bilateral knee, bilateral
hand-wrist, anteroposterior pelvis, bilateral lumbosacral,
posteroanterior lung which includes bilateral shoulder
joints and bilateral skull. We have discovered that speci-
fied lesions are symmetrically distributed around bilat-
eral hand-wrist (Figure 1(a)), bilateral foot-ankle (Fig-
ure 1(b)), bilateral knee joint (Figure 1(c)), bilateral
shoulder joint (Figure 1(d)) and bilateral hip joint (Fig-
ure 1(e)). Ankle magnetic resonance (Figures 2(a)-(b))
examination carried out on the external centre indicated
that lesions having similar features were monitored on
bone structures. The patient did not have active com-
plaints about locations which correspond to lesions as in-
dicated in direct radiographs. Complete blood count, al-
kene phosphatase, serum calcium, phosphor and magne-
sium levels, parathyroid hormone, erythrocyte sedimen-
tation rate, C reactive protein, liver and kidney function
tests and D vitamin levels were normal. Bone mineral
densitometry measures of the patient were within normal
levels. TSH level of the patient was high and free T4
level was low, and the patient was referred to endocrine
department for consultation. As a result of the consulta-
tion, the patient was recommended thyroid hormone
*Corresponding author.
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Bone Pathology: Osteopoikilosis
218
(a) (b)
(c) (d)
(e)
Figure 1. (a) Bilateral hand-wrist; (b) Foot-ankle; (c) Bilat-
eral knee joint; (d) Bilateral shoulder joint; (e) Bilateral hip
joint.
treatment by the endocrine department. The department
did not provide additional recommendations for bone
pathology. We prescribed orthopaedic follow-up for the
patient due to the current situation.
3. Discussion
Osteopoikilosis is a rare benign sclerotic bone dysplasia.
Autosomal dominant inheritance is observed and the rea-
son of the pathology is unknown [1]. The disease which
usually develops asymptomatically is coincidentally di-
agnosed with direct radiographs [2]. The patients are ge-
nerally asymptomatic, yet pain can be a rare symptom of
the disease [3]. Characteristically, the disease is observed
in direct radiographs as multiple oval and round densities
have 2 - 10 mm size on epiphyseal and metaphyseal re-
gions on long bones [4]. The studies indicate that the
disease is inherited, and has autosomal dominant inheri-
tance [4-6]. Patients with osteopoikilosis should be fol-
lowed up with regard to complications such as malignant
transformation, spinal stenosis and hip fracture although
the course of disease is benign [2]. During differential
diagnosis, osteoblastic metastases, tuberous sclerosis,
(a)
(b)
Figure 2. Ankle magnetic resonance.
mastocytosis, osteopathy, striata, melorheostosis must be
taken into consideration. Osteopoikilosis can be distin-
guished by symmetrical distribution, metaphyseal and
epiphyseal involvement and uniform size of the lesions
[1]. Lesions descried on our subject were observed to be
symmetrical with metaphyseal and epiphyseal involve-
ment. In conclusion, patients with osteopoikilosis remain
asymptomatic and diagnosed incidentally on direct ra-
diographs take separates from other diseases. Patients
with osteopoikilosis must be advised that the disease has
benign course, and similar pathologies can be found with
their family members, and they must be followed-up for
possible complications.
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