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Open Access Case Report

Two cases of multiple ossifying fibromas in the jaws

Ting-Ting Wang1, Ran Zhang2, Lin Wang1*, Yan Chen2, Qing Dong1 and Tie-Jun Li234*

Author Affiliations

1 Department of Oral Medicine, Hebei United University, School and Hospital of Stomatology, Tangshan, 82 South Construction Road, Hebei 063000, Lubei District, PR China

2 Department of Oral Pathology, Peking University School and Hospital of Stomatology, 22 South Zhongguancun Avenue, Haidian District, Beijing 100081, PR China

3 Central Laboratory, Peking University School and Hospital of Stomatology, Beijing, China

4 National Engineering Laboratory for Digital and Material Technology of Stomatology, Beijing, China

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Diagnostic Pathology 2014, 9:75  doi:10.1186/1746-1596-9-75

Published: 28 March 2014

Abstract

Background

The clinicopathologic characteristics of multiple ossifying fibroma (OF) are unclear due to the condition’s rarity, making diagnosis challenging. Sporadic multiple OFs must be distinguished from hyperparathyroidism-jaw tumour syndrome (HPT-JT) related OF and other fibro-osseous lesions.

Methods

Multiple OF cases were identified from ossifying fibroma cases. Clinical data including age, sex, anatomic site, radiographic features, clinical impression, treatment and available follow-up data as well as serum calcium, phosphorus, and parathyroid hormone (PTH) were recorded. GNAS and HRPT2 genetic mutations were examined in the two present cases. Case reports of sporadic multiple ossifying fibroma and HPT-JT-related OF were also reviewed.

Results

The two present cases were confirmed as sporadic multiple OF, with no genetic GNAS and HRPT2 mutations found. The incidence of sporadic multiple ossifying fibroma was 2.0% (2/102). The total 18 sporadic multiform OF cases were characterized as followed: 13 (72.2%) female; 5 (27.8%) male; mean age 28.6 years; 2/16 (11.1%) cases only in the mandible; 4/18 (22.2%) cases only in the maxilla; and 12/18 (66.7%) cases in both the maxilla and mandible. Radiographically, the lesions were radiolucent in 5/18 (27.8%) cases and mixed density in 13/18 (72.2%) cases. Along with 24 cases of HPT-JT related OF were reviewed, sixteen (66.7%) patients were diagnosed with a single lesion, and 8 patients (33.3%) were diagnosed with multiple jaw lesions.

Conclusions

Sporadic multiple OFs are very rare, but must be distinguished from HPT-JT related OF. We strongly recommend that patients diagnosed with multiple ossifying fibromas receive serum PTH testing and mutation screening of HRPT2.

Virtual slides

http://www.diagnosticpathology.diagnomx.eu/vs/1194507146115753 webcite

Keywords:
Multiple ossifying fibroma; HPT-JT; Fibrous dysplasia; GNAS gene; HRPT2 gene; Osseous dysplasia