Giant peripheral osteoma of the mandible.
Abstract: Osseous expansion of any body part is an unwelcome guest and deep are its impacts when it is located on the face. The bigger the lesion, the more bitter is the psychosocial trauma to the affected individual. This article describes the case of a 50 year old female who presented with painless swelling of the right submandibular region manifesting as a dreadful cosmetic disfigurement. The mass had been progressing slowly for the last 15 years. Imaging showed a giant peripheral osteoma of 10.8 cm involving buccal and lingual surface of the body, ramus, angle and inferior border of the right side of mandible. To the best of our knowledge, a giant peripheral osteoma of mandible having size more than 10 cm has never been reported earlier.

KEY WORDS: Giant peripheral osteoma; Swelling of mandible; CT scan, Panoramic view
Article Type: Case study
Subject: Osteoma (Diagnosis)
Osteoma (Care and treatment)
Osteoma (Research)
Biopsy, Needle (Usage)
Authors: Kachewar, Sushil
Bhadane, Sushant
Kulkarni, Devidas
Sankaye, Smita
Pub Date: 01/01/2012
Publication: Name: Internet Journal of Medical Update Publisher: Dr. Arun Kumar Agnihotri Audience: Academic; Professional Format: Magazine/Journal Subject: Health Copyright: COPYRIGHT 2012 Dr. Arun Kumar Agnihotri ISSN: 1694-0423
Issue: Date: Jan, 2012 Source Volume: 7 Source Issue: 1
Topic: Event Code: 310 Science & research
Geographic: Geographic Scope: India Geographic Code: 9INDI India
Accession Number: 277601648
Full Text: INTRODUCTION

Benign bony outgrowths of the compact or the cancellous bones result in formation of an osteoma. Only those arising from periosteum are labelled as peripheral osteomas and have varied etiology like being a true neoplasm, post traumatic entity, post infective or as an entity arising due to muscle traction. They are slowly growing, clinically silent and usually manifest only as focal asymmetry or disfigurement.

Constellation of osteomas, supernumerary and or impacted teeth, adenomatoid gastrointestinal polyps and tumours of soft tissues and skin; constitutes Gardner s syndrome. Such polyps in gastrointestinal system usually turn malignant. Diagnosing osteoma is therefore important and has a life saving potential because they can be the earliest presenting feature of Gardner's syndrome. This case report not only describes the largest peripheral osteoma in the literature available till date, but also shows how disfiguring an unattended and neglected benign facial osseous lesion can turn. Initially asymptomatic but later ghastly psychosocially traumatic facial bone osteoma therefore calls for prompt surgical action and a timely nip in the bud. The role of imaging is important as it aids in diagnosing and managing the osteoma per se, and can rule in or rule out the possible existence and /or evolution of Gardner's syndrome. Osteomas from mandible are rare; and rarer still is a giant osteoma from mandible. Hence it is being reported here. Relevant clinical data from the earlier published literature and the main differential diagnoses are also discussed.

CASE REPORT

A 50 year old female presented with painless swelling and resultant cosmetic disfigurement of right submandibular region (Figure 1a & b). There was no history of any local trauma. The mass had been progressing slowly for the last 15 years. On examination, a subcutaneous painless bony-hard mass in the submandibular area was palpable. Lateral mandibular radiograph (Figure 2a) and Panoramic view (Figure 2b) revealed a large exophytic radiodense mass arising from right side of the mandible. Axial (Figure 3a) and reformatted coronal (Figure 3b) CT scan bone window images demonstrated a large lobulated, bony mass attached to the buccal and lingual surface of the body, ramus, angle and inferior border of right mandible. The mass protruded towards the infero-lateral surface of the mandible and caused medial displacement of the right submandibular gland. Superiorly the mass extended up to the mandibular notch and closely abutted the lateral pterygoid muscle. The bony mass was of the compact type and measured 10.8 x 4.0 x 4.6 cm. The clinical and radiographic findings were consistent with giant osteoma.

[FIGURE 1 OMITTED]

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Fine needle aspiration cytology (Figure 4) showed typical features of osteoma that confirmed the diagnosis. Diagnostic colonoscopy did not reveal any colonic polyps. The patient was referred to a higher centre for cosmetic corrective surgery. Unfortunately, the patient did not return for follow up.

[FIGURE 4 OMITTED]

DISCUSSION

Osteoma is a benign bone tumor with origins reported from periosteum, endosteum and even from extra-skeletal soft tissue. Central osteomas arise from endosteum and peripheral osteomas arise from periosteum. Although the exact cause is yet unknown, osteomas are formed when there is relentless unchecked growth in a bone (1). Hence, it is still a dilemma whether to call it a true neoplasm, developmental anomaly; or a reactive response to infection, trauma or muscle traction (2). Peripheral osteomas are located in close proximity to muscle attachment (i.e. masseter, medial pterygoid, temporalis) (3). Because of their slow growth, majority of the affected individuals have no symptoms. Hence they are noticed by patients only when they cause visible swelling or disfigurement and feel hard on palpation (1). Young adults are the commonest victims of mandibular osteomas in whom they manifest as umbrella shaped lesions and have a stalk. On radiographs, they appear as lobulated opacities. Histological confirmation is a must to further grade them into a compact, cancellous or mixed variety of peripheral osteoma (4). Dental and skeletal abnormalities when associated with colorectal polyposis constitutes Gardner's syndrome. Relevant investigations must therefore be done to evaluate patients along this line (4). Osteomas can occur in any bone but are found more frequently in the skull, ethmoid sinuses, mandible, and maxilla (5).

This is a case of giant osteoma involving buccal and lingual surface of the body, ramus, angle and inferior border of the right mandible. It was a well circumscribed slow-growing hard mass and produced disfigurement. Sometimes osteoma can cause functional impairment such as difficulty in chewing, dysphagia and limited mandibular movement. Large peripheral osteomas of size up to 87 mm have been reported (6). In this case, size of mass lesion was 108 mm. Absence of colorectal polyps, skeletal abnormalities and multiple impacted or supernumerary teeth in this case, was against labelling it as Gardner's syndrome. Table 1 shows the summary of clinical and radiological features present in our case and some recent studies (7-11).

CONCLUSION

Although peripheral osteomas are usually small in size, this case demonstrates that when neglected they can slowly enlarge considerably. Cosmetic disfigurement and functional impairment brings the patient for treatment. However it is expected of a vigilant medical professional that he actively rule out the possibility of Gardner's syndrome as a simple osteoma could be the earliest sign that heralds this syndrome which ultimately manifests as malignant polyps in gastrointestinal system.

REFERENCES

(1.) Woldenberg Y, Nash M, Bodner L. Peripheral osteoma of the maxillofacial region. Diagnosis and management: a study of 14 cases. Med Oral Patol Oral Cir Bucal. 2005 Jul;10 Suppl 2:E139-42.

(2.) Kerckhaert A, Wolvius E, van der Wal K, et al. A giant osteoma of the mandible: case report. J Craniomaxillofac Surg. 2005 Aug;33(4):282-5.

(3.) Lt Col Roy ID. Peripheral osteoma of mandible. MJAFI. 2008;64:385-6.

(4.) Richardson PE, Arendt DM, Fidler JE, et al. Radiopaque mass in the submandibular region. J Oral Maxillofac Surg. 1999 Jun;57(6):709-13.

(5.) Sayan NB, Ucok C, Karasu HA, et al. Peripheral osteoma of the oral and maxillofacial region: a study of 35 new cases. J Oral Maxillofac Surg. 2002 Nov;60(11):1299-301.

(6.) Seo YA, Chang HA, Karp SC. Giant osteoma of the mandible causing breathing problem. Korean J Oral Maxillofac Radiol. 2006;36:217-20.

(7.) Bulut E, Acikgoz A, Ozan B et al. Large peripheral osteoma of the mandible: a case report. Int J Dent. 2010;2010:834761. Epub 2011 Feb 6.

(8.) Goudar G, RaviKumar R, Manjunath GA, et al. Osteoma of the mandible. J Dent Sci Res. 2011 Feb;2(1):116-22.

(9.) Kaya GS, Omezli MM, Sipal S, et al. Oral myiasis: gigantic peripheral osteoma of the mandible: a case report. J Clin Exp Dent. 2010;2(3):e160-2.

(10.) Donohue-Cornejo A, Franco Garrocho LE, Albarran-Vergara S, et al. Active Giant Peripheral Osteoma of the Mandible. Presentation of one case with follow-up to 6 years. J Clin Exp Dent. 2010;2(4):e212-4.

(11.) Bhuvaneshwari S, Ahmed J, Goel S, et al. Peripheral osteoma-a case report. JMBS. 2010 Nov;1(3)31-6.

Sushil Kachewar * ([PSI]) MD, DNB, Sushant Bhadane ** MD, Devidas Kulkarni ([dagger]) MD and Smita Sankaye ([double dagger]) MBBS

* Associate Professor, ([dagger]), Department of Radiology, ([double dagger]) Resident, Department of Pathology, Rural Medical College, Loni, Maharashtra, India ** Consultant Radiologist, Nashik, Maharashtra, India

(Received 09 March 2011 and accepted 01 May 2011)

([PSI]) Correspondence at: Associate Professor, Department of Radiology, Rural Medical College, Pravara Medical Trust, PIMS (DU), Loni 413736, Maharashtra, India; Mobile: 0091-9921160357; Email: sushilkachewar@hotmail.com
Table 1: Comparative analysis of features of mandible peripheral
osteomas from some published studies

              Bulut et   Goudar et   Kaya et         Donohue-
Criteria       al (7)     al (8)      al (9)    Cornejo et al (10)

Age (yrs)        37         29          55              17

Sex              F           M          F               F

Present          6          12          ?               10
since (yrs)

Symptoms         -           -          -               -

Deformity        +           +          +               +

Size in cm       3           3          -              4.2

Labelled as    Large         -       Gigantic         Giant

Radiopaque       +           +          +               +
mass

Stalk            +           -          -               ?

Surgical         +           +          +               +
Excision

              Bhuvaneshwari   Present
Criteria       et al (11)      Study

Age (yrs)          30           50

Sex                 F            F

Present             3           15
since (yrs)

Symptoms            -            -

Deformity           +            +

Size in cm         1.2         10.8

Labelled as         -          Giant

Radiopaque          +            +
mass

Stalk               +            -

Surgical            +            +
Excision
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