Giant osteoid osteoma of tibial shaft: A rare case report

Case Report

Giant osteoid osteoma of tibial shaft: A rare casereport

Raju Vaishya a,*, Shameem Ahmad Khan b, Ashok Kumar c

a Prof., Sr Consultant, Department of Orthopaedics, Indraprastha Apollo Hospitals, New Delhi 110076, IndiabRegistrar, Department of Orthopaedics, Indraprastha Apollo Hospitals, New Delhi 110076, IndiacOrtho OT Nurse, Department of Orthopaedics, Indraprastha Apollo Hospitals, New Delhi 110076, India

a r t i c l e i n f o

Article history:

Received 20 July 2012

Received in revised form

11 August 2012

Accepted 13 August 2012

Available online 27 August 2012

Keywords:

Giant osteoid osteoma

Osteoblastoma

Diaphyseal

Non-steroidal anti inflammatory

drugs

a b s t r a c t

We report a rare case of diaphyseal, giant osteoid osteoma of tibial shaft. Detailed review of

literature of giant osteoid osteoma is presented. This entity is more clearly defined and its

differentiating features with other mimicking lesions are presented.

Copyright ª 2012, Indraprastha Medical Corporation Ltd. All rights reserved.

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 Inflam-

matory 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).

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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 ante-

rior cortex of the mid shaft of the left tibia with slightly ill

definedmargins 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 perile-

sional 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 radi-

ography, intra-operatively using image intensifier. The tumor

was excised en bloc (Fig. 3). A bony hard tissue of about

7 � 3 � 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 � 3 � 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 � 1.4 � 0.5 cm (Fig. 4).

The microscopic examination showed sections from cen-

ter of the bone trabeculae of variably mineralized osteoid.

Most of these have a prominent osteoblastic rimming. The

intervening stroma is composed of loose fibroconnective tis-

sue, with prominent vascularity. Areas of fresh & old hemor-

rhage 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 evi-

dence of recurrence clinically or radiologically. The main pre-

operative complaint of persistent pain resolved completely,

post-operatively, without any need for any analgesics.

Discussion

Gaint osteoid osteoma was first described as an osteoblastic-

osteoid tissue-forming tumor by Jaffe and Mayer in 1932.1

Subsequently, Lichtenstein2 reported similar lesions as oste-

ogenic fibromas and Dahlin and Johnson,3 reported them as

giant osteoid osteomas (distinguishing them from ossifying

fibromas and classical osteoid osteomas). The term osteo-

blastoma 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

Fig. 4 e Tumor removed en bloc.

Table 1 e Differential diagnosis of benign diaphyseal tumors of tibia.

Osteoblastoma 10e30 Spine,

femur &

tibia shaft

Dull pain,

scoliosis,

neuro deficit

(spine)

>2 cm in size,

osteolytic

lesion þ/�nidus, with

sclerosis

Fibro-vascular

stroma, primitive

woven bone, layer

of osteoblasts

En bloc resec-

tion, extended

curettage þ/�bone grafting

Usually

benign

Osteoid

osteoma

10e30 Femur, tibia Night pain

relived with

NSAID

Central nidus

(<1.5 cm) with

surrounding

sclerosis

Fibro-vascular

tissue with imma-

ture bone

Excision, curet-

tage, percuta-

neous RF

ablation

Always

benign, self

limiting

condition

Osteosarcoma 10e25 Femur. tibia

(around

knee)

Pain, swelling,

malignancy

signs

Aggressive

metaphyseal

lesion e blastic/

lytic

Malignant osteoid,

pleomorphic oste-

oblasts in multiple

layers

Wide resection,

amputation

Highly malig-

nant, early

pulmonary

metastasis

Giant cell

tumor (GCT)

20e40 Around

knee, distal

radius

Swelling Eccentric, epiph-

yseal osteolytic

lesion

Multinucleated

giant cells in back-

ground of stromal

cells

Excision,

extended

curettage þ/�cementing

Rarely malig-

nant, locally

aggressive

Eosinophilic

granuloma

05e20 Spine, long

bone

(diaphysis)

Back pain Vertebra plana,

punched out

lesion

Langerhan’s cell,

eosinophilic

cytoplasm

Low-dose irradi-

ation, curettage

& bone grafting

Self limiting

Adamantinoma 15e30 Tibial shaft

(85%),

mandible

Pain,

swelling

Multiple, sharply

demarcated

radiolucent

lesions

Islands of epithelial

cells in a fibrous

stroma

Wide resection

or amputation

Radio/chemo

therapy

resistant

Brodie’s

abscess

15e25 Metaphysis

around

knee

Dull aching

pain

Lytic lesion with

a rim of sclerosis

Infected granulation

tissue

Curettage/

saucerization

Low virulent

organism

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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 excru-

ciating 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 osteo-

lytic 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

andmay be surrounded by variable amounts of sclerotic bone.

On cut section the lesion appears friable, gritty and hemor-

rhagic. 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 trabec-

ulae 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 Lich-

tenstein6 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.

r e f e r e n c e s

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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