Giant osteoid osteoma of the tibial shaft is a rare entity. Though this tumor is seen commonly in axial skeleton, so far no conclusive report has been published on its periosteal involvement of tibial shaft diaphysis.

1. Giant osteoid osteoma of tibial shaft: A rare case report

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Case Report
Giant osteoid osteoma of tibial shaft: A rare case
report
Raju Vaishya a,*, Shameem Ahmad Khan b, Ashok Kumar c
a
Prof., Sr Consultant, Department of Orthopaedics, Indraprastha Apollo Hospitals, New Delhi 110076, India
Registrar, Department of Orthopaedics, Indraprastha Apollo Hospitals, New Delhi 110076, India
c
Ortho OT Nurse, Department of Orthopaedics, Indraprastha Apollo Hospitals, New Delhi 110076, India
b
article info
abstract
Article history:
We report a rare case of diaphyseal, giant osteoid osteoma of tibial shaft. Detailed review of
Received 20 July 2012
literature of giant osteoid osteoma is presented. This entity is more clearly defined and its
Received in revised form
differentiating features with other mimicking lesions are presented.
11 August 2012
Copyright ª 2012, Indraprastha Medical Corporation Ltd. All rights reserved.
Accepted 13 August 2012
Available online 27 August 2012
Keywords:
Giant osteoid osteoma
Osteoblastoma
Diaphyseal
Non-steroidal anti inflammatory
drugs
Introduction
Giant osteoid osteoma of the tibial shaft is a rare entity.
Though this tumor is seen commonly in axial skeleton, so far
no conclusive report has been published on its periosteal
involvement of tibial shaft diaphysis. The lesion generally
produces few symptoms in spite of its relatively large size.
Radiologically, it presents as a lytic lesion of bone; however,
varying degrees of calcification and peripheral sclerosis may
give it a bizarre appearance. Its awareness is necessary as it
may be confused clinically with other benign and malignant
tumors of the bone. This benign bone tumor requires local
excision as its definitive treatment.1
Case report
A 16 year old boy presented with a 2 years history of dull
aching pain (intermittent) and swelling of left middle 3rd leg.
There was temporary relief with Non-Steroidal Anti Inflammatory Drugs (NSAIDs). There was no other associated
symptoms or family history of similar problem.
* Corresponding author. Tel.: þ91 9810123331.
E-mail address: raju.vaishya@gmail.com (R. Vaishya).
0976-0016/$ e see front matter Copyright ª 2012, Indraprastha Medical Corporation Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.apme.2012.08.006

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On examination, he has had a bony hard, tender swelling
of about 6 cm  5 cm present on the anterior aspect of middle
third of left leg, seems to be arising (and attached) from the
tibial shaft. Local temperature over the swelling was slightly
raised. Ipsilateral ankle, knee and hip movements were
normal and there was no other abnormal swellings found in
other parts of the body.
All the laboratory parameters were within normal limits,
including ESR and CRP.
X-rays, showed an eccentric radiolucent area in the anterior cortex of the mid shaft of the left tibia with slightly ill
defined margins and a surrounding area of dense sclerosis and
solid periosteal reaction involving the cortex of the bone and
some scalloping of the anterior tibial cortex intramedullary
(but no extension into it), due to pressure effect of the bony
mass (Fig. 1).
Computed Tomographic (CT) scan showed an osteolytic
diaphyseal cortical-based lesion in the anterior cortex of the
left tibia with sparse intralesional trabeculations and perilesional osteosclerosis. Hyperdense foci are also noted in the
adjoining part of medullary cavity (Fig. 2).
A wide excision and biopsy of the lesion was done by an
anterior approach. The lesion was demarcated well by radiography, intra-operatively using image intensifier. The tumor
was excised en bloc (Fig. 3). A bony hard tissue of about
7 Â 3 Â 2 cm was excised. It was arising from the periosteal
surface of the anterior cortex of mid shaft tibia.
On gross examination, there was a single, flat bony piece of
tissue, measuring 6.5 Â 3 Â 1.5 cm. Cut surface show central,
Fig. 1 e Pre-op X-rays.
Fig. 2 e Pre-op CT scan.
Fig. 3 e Post-op X-rays showing en bloc resection of tumor.

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irregular, soft, dark brown cavitated area, roughly measuring
3 Â 1.4 Â 0.5 cm (Fig. 4).
The microscopic examination showed sections from center of the bone trabeculae of variably mineralized osteoid.
Most of these have a prominent osteoblastic rimming. The
intervening stroma is composed of loose fibroconnective tissue, with prominent vascularity. Areas of fresh & old hemorrhage are seen. Sections from the periphery show broad
trabeculae of mature lamellar bone. The histopathology was
considered consistent with a giant osteoid osteoma.
Gram and AFB stains with Aerobic, Anaerobic and Fungal
cultures were negative.
The wound healed by primary intention. The patient was
mobilized non-weight bearing for 3 weeks with crutches. At 2
years follow-up the patient had no pain, swelling or any evidence of recurrence clinically or radiologically. The main preoperative complaint of persistent pain resolved completely,
post-operatively, without any need for any analgesics.
Discussion
Fig. 4 e Tumor removed en bloc.
Gaint osteoid osteoma was first described as an osteoblasticosteoid tissue-forming tumor by Jaffe and Mayer in 1932.1
Subsequently, Lichtenstein2 reported similar lesions as osteogenic fibromas and Dahlin and Johnson,3 reported them as
giant osteoid osteomas (distinguishing them from ossifying
fibromas and classical osteoid osteomas). The term osteoblastoma was introduced by Jaffe4 in 1956 and the prefix
“benign” was added to stress its benign nature, in contrast to
osteogenic sarcoma with which it is frequently confused.
Benign osteoblastoma is a very uncommon lesion. Males and
Table 1 e Differential diagnosis of benign diaphyseal tumors of tibia.
Osteoblastoma
10e30
Spine,
femur &
tibia shaft
Dull pain,
scoliosis,
neuro deficit
(spine)
Osteoid
osteoma
10e30
Femur, tibia
Night pain
relived with
NSAID
Osteosarcoma
10e25
Femur. tibia
(around
knee)
Pain, swelling,
malignancy
signs
Swelling
Giant cell
tumor (GCT)
20e40
Around
knee, distal
radius
Eosinophilic
granuloma
05e20
Back pain
Adamantinoma
15e30
Spine, long
bone
(diaphysis)
Tibial shaft
(85%),
mandible
Brodie’s
abscess
15e25
Metaphysis
around
knee
Dull aching
pain
Pain,
swelling
>2 cm in size,
osteolytic
lesion þ/À
nidus, with
sclerosis
Central nidus
(<1.5 cm) with
surrounding
sclerosis
Aggressive
metaphyseal
lesion e blastic/
lytic
Eccentric, epiphyseal osteolytic
lesion
Vertebra plana,
punched out
lesion
Multiple, sharply
demarcated
radiolucent
lesions
Lytic lesion with
a rim of sclerosis
Fibro-vascular
stroma, primitive
woven bone, layer
of osteoblasts
En bloc resection, extended
curettage þ/À
bone grafting
Usually
benign
Fibro-vascular
tissue with immature bone
Excision, curettage, percutaneous RF
ablation
Wide resection,
amputation
Always
benign, self
limiting
condition
Highly malignant, early
pulmonary
metastasis
Rarely malignant, locally
aggressive
Malignant osteoid,
pleomorphic osteoblasts in multiple
layers
Multinucleated
giant cells in background of stromal
cells
Langerhan’s cell,
eosinophilic
cytoplasm
Islands of epithelial
cells in a fibrous
stroma
Infected granulation
tissue
Excision,
extended
curettage þ/À
cementing
Low-dose irradiation, curettage
& bone grafting
Wide resection
or amputation
Self limiting
Curettage/
saucerization
Low virulent
organism
Radio/chemo
therapy
resistant

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females are affected with equal frequency. The majority of
cases occur between 10 and 35 years of age. The youngest
patient reported was 5 years, the oldest was 61 years.
Although the lesion may involve any bone, the vertebrae,
femur and tibia are most commonly affected.
Characteristically, giant osteoid osteomas grows to a large
size, yet produces few or no symptoms. This is in contrast to
osteoid osteoma which is usually small but produces excruciating pain. We believe that this may be explained on the
basis that the nidus in a small osteoid osteoma is densely
encapsulated by thick bone & not allowing it to expand
(‘breathe’), causing severe pain. Whereas, in a giant osteoid
osteoma, since the nidus has larger space to expand, there is
lesser degree of pain.
As a rule, laboratory tests are within normal limits. The
radiologic appearance is that of a well-circumscribed osteolytic lesion. The overlying cortex may be thinned or eroded.
Depending on the extent of calcification within the tumor,
varying degrees of sclerosis are apparent. Although sclerosis
about a central nidus may occur, it is seldom as characteristic
as in osteoid osteoma. The tumor may vary from 2 to 12 cm in
greatest diameter. It appears pinkish-red to purple in color
and may be surrounded by variable amounts of sclerotic bone.
On cut section the lesion appears friable, gritty and hemorrhagic. Microscopically, osteoid trabeculae are seen lying in
loose vascular osteoblastic connective tissue. The osteoid
trabeculae are lined by typical osteoblasts which show no
evidence of malignancy. Mitoses are rare, thick bony trabeculae may be present at the periphery. Multinucleated giant
cells, probably osteoclasts, are also present in variable
numbers, and evidence of remote hemorrhage is frequently
seen in the poorly cellular connective tissue. Cartilage is never
present.
Giant osteoid osteoma may be confused with other
mimicking lesions of the bone, as discussed in the Table 1
below:
According to Dahlin and Johnson,3 “the lesion is essentially
osteoid osteoma, but fails to demonstrate aggressiveness”.
This concept is also advanced by Lichtenstein,3 who regards
osteoma and osteoid osteoma as special types of benign
osteoblastoma. The current belief that these lesions represent
a primary benign bone tumor was proposed by Jaffe1 in 1935.
Prior to that time a non-bacterial inflammatory origin was
considered. Recently, there have been several reports
describing clinical and roentgenographic healing, and the
validity of the classification of this lesion as a neoplasm has
been challenged.5 Since both osteoid osteoma and benign
osteoblastoma show characteristic osteoblastic proliferation
and osteoid formation in a highly vascular stroma, it is
conceivable that a locally altered blood supply (for reasons not
readily apparent) stimulates osteoblastic activity and results
in either lesion. Such a pathogenesis was proposed by Lichtenstein6 for the development of aneurysmal bone cyst, which
has a similarly prominent vasculature. This concept may well
explain the good results of conservative surgical therapy e
local excision or curettage.7
Conflicts of interest
All authors have none to declare.
references
1. Jaffe HL. Arch Surg (Chicago). 1935;31:709.
2. Lichtenstein L. Bone Tumors. St. Louis: C. V. Mosby Company;
1952. p. 82.
3. Dahlin DC, Johnson Jr EW. J Bone Joint Surg Am. 1954;36A:559.
4. Dahlin DC, Johnson Jr EW. Bull Hosp Jt Dis. 1956;17:141.
5. Moberg E. J Bone Joint Surg Am. 1951;33A:166.
6. Moberg E. Bone Tumors. 2nd ed. St. Louis: C. V. Mosby Company;
1959. p. 97.
7. Ochsner Sr A, Ochsner Jr A. Tumors of the thoracic wall. In:
Spain DM, ed. Diagnosis and Treatment of Tumors of the Chest.
New York: Grune & Stratton, Inc.; 1960:205.

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