Top

Diagnostic Pathology

Published in:

Open Access 01-12-2014 | Case Report

Two cases of multiple ossifying fibromas in the jaws

Authors: Ting-Ting Wang, Ran Zhang, Lin Wang, Yan Chen, Qing Dong, Tie-Jun Li

Published in: Diagnostic Pathology | Issue 1/2014

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

Available only for authorised users

Eversole R, Su L, ElMofty S: Benign fibro-osseous lesions of the craniofacial complex: a review. Head Neck Pathol. 2008, 2: 177-202. 10.1007/s12105-008-0057-2.PubMedPubMedCentralCrossRef

Barnes L, Eveson J, Reichart P, Sidransky D: Book World Health Organization Classification of Tumours: Pathology and Genetics of Head and Neck Tumours. World Health Organization Classification of Tumours: Pathology and Genetics of Head and Neck Tumours. 2005, Lyon: IARC Press

Waldron CA: Fibro-osseous lesions of the jaws. J Oral Maxillofac Surg. 1993, 51: 828-835. 10.1016/S0278-2391(10)80097-7.PubMedCrossRef

Chambers MS, Rassekn CH, Toth BB, Lemon JC, Hoffman RD: A maxillary fibro-osseous lesion: differential diagnosis and case report. Tex Dent J. 2002, 119: 12-19.PubMed

Toyosawa S, Yuki M, Kishino M, Ogawa Y, Ueda T, Murakami S, Konishi E, Iida S, Kogo M, Komori T, Tomita Y: Ossifying fibroma vs fibrous dysplasia of the jaw: molecular and immunological characterization. Modern Pathol. 2007, 20: 389-396. 10.1038/modpathol.3800753.CrossRef

Alawi F: Benign fibro-osseous diseases of the maxillofacial bones. A review and differential diagnosis. Am J Clin Pathol. 2002, 118 (Suppl): S50-S70.PubMed

Kusano T, Hirabayashi S, Eguchi T, Sugawara Y: Treatment strategies for fibrous dysplasia. J Craniofac Surg. 2009, 20: 768-770. 10.1097/SCS.0b013e3181a14ca8.PubMedCrossRef

Slootweg PJ: Bone diseases of the jaws. Int J Dent. 2010, 2010: 702314-PubMedPubMedCentralCrossRef

Shi RR, Li XF, Zhang R, Chen Y, Li TJ: GNAS mutational analysis in differentiating fibrous dysplasia and ossifying fibroma of the jaw. Modern Pathol. 2013, 26: 1023-1031. 10.1038/modpathol.2013.31.CrossRef

10.

Brannon RB, Fowler CB: Benign fibro-osseous lesions: a review of current concepts. Adv Anat Pathol. 2001, 8: 126-143. 10.1097/00125480-200105000-00002.PubMedCrossRef

11.

Kuznetsov SA, Cherman N, Riminucci M, Collins MT, Robey PG, Bianco P: Age-dependent demise of GNAS-mutated skeletal stem cells and “normalization” of fibrous dysplasia of bone. J Bone Miner Res. 2008, 23: 1731-1740. 10.1359/jbmr.080609.PubMedPubMedCentralCrossRef

12.

Aldred MJ, Talacko AA, Savarirayan R, Murdolo V, Mills AE, Radden BG, Alimov A, Villablanca A, Larsson C: Dental findings in a family with hyperparathyroidism-jaw tumor syndrome and a novel HRPT2 gene mutation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006, 101: 212-218. 10.1016/j.tripleo.2005.06.011.PubMedCrossRef

13.

Cavaco BM, Barros L, Pannett AA, Ruas L, Carvalheiro M, Ruas MM, Krausz T, Santos MA, Sobrinho LG, Leite V, Thakker RV: The hyperparathyroidism-jaw tumour syndrome in a Portuguese kindred. QJM. 2001, 94: 213-222. 10.1093/qjmed/94.4.213.PubMedCrossRef

14.

Jackson CE, Norum RA, Boyd SB, Talpos GB, Wilson SD, Taggart RT, Mallette LE: Hereditary hyperparathyroidism and multiple ossifying jaw fibromas: a clinically and genetically distinct syndrome. Surgery. 1990, 108: 1006-1012. discussion 1012–1003PubMed

15.

Yamashita Y, Akiyama T, Mizusawa N, Yoshimoto K, Goto M: A case of hyperparathyroidism-jaw tumour syndrome found in the treatment of an ossifying fibroma in the maxillary bone. Int J Oral Max Surg. 2007, 36: 365-369. 10.1016/j.ijom.2006.08.007.CrossRef

16.

Carpten J, Robbins C, Villablanca A, Forsberg L, Presciuttini S, Bailey-Wilson J, Simonds W, Gillanders E, Kennedy A, Chen J: HRPT2, encoding parafibromin, is mutated in hyperparathyroidism–jaw tumor syndrome. Nat Gen. 2002, 32: 676-680. 10.1038/ng1048.CrossRef

17.

Shattuck TM, Valimaki S, Obara T, Gaz RD, Clark OH, Shoback D, Wierman ME, Tojo K, Robbins CM, Carpten JD, Farnebo LO, Larsson C, Arnold A: Somatic and germ-line mutations of the HRPT2 gene in sporadic parathyroid carcinoma. New Engl J Med. 2003, 349: 1722-1729. 10.1056/NEJMoa031237.PubMedCrossRef

18.

Pimenta FJ, Gontijo Silveira LF, Tavares GC, Silva AC, Perdigao PF, Castro WH, Gomez MV, Teh BT, De Marco L, Gomez RS: HRPT2 gene alterations in ossifying fibroma of the jaws. Oral Oncol. 2006, 42: 735-739. 10.1016/j.oraloncology.2005.11.019.PubMedCrossRef

19.

Haven CJ, Wong FK, van Dam EW, van der Luijt R, van Asperen C, Jansen J, Rosenberg C, de Wit M, Roijers J, Hoppener J: A genotypic and histopathological study of a large Dutch kindred with hyperparathyroidism-jaw tumor syndrome. J Clin Endocr Metab. 2000, 85: 1449-1454.PubMed

20.

Hobbs MR, Pole AR, Pidwirny GN, Rosen IB, Zarbo RJ, Coon H, Heath H, Leppert M, Jackson CE: Hyperparathyroidism-jaw tumor syndrome: the HRPT2 locus is within a 0.7-cM region on chromosome 1q. Am J Hum genet. 1999, 64: 518-525. 10.1086/302259.PubMedPubMedCentralCrossRef

21.

Popli DB, Desai R, Bansal S, Andrade NN: Bilateral psammomatoid ossifying fibroma: a case report and review of the literature. J Oral Maxillofac Surg. 2013, 71: 714-720. 10.1016/j.joms.2012.10.010.PubMedCrossRef

22.

Desai K, Gupta K, Manjunatha BS, Palan S: Bimaxillary presentation of central ossifying fibroma: a unique aggressive entity. BMJ Case Rep. 2013, 2013: doi:10.1136/bcr-2013-010124

23.

Ponniah I, Sethurajan SB, Rajiah D: A multilocular radiolucency with spindle cell proliferation in a case of ossifying fibroma: a potential pitfall. Dento Maxillo Facial Radiol. 2012, 41: 605-608. 10.1259/dmfr/28249452.CrossRef

24.

Kutcher MR, Rigby MH, Bullock M, Trites J, Taylor SM, Hart RD: Hyperparathyroidism-jaw tumor syndrome. Head Neck. 2013, 35: E175-E177. 10.1002/hed.22918.PubMedCrossRef

25.

Iacobone M, Masi G, Barzon L, Porzionato A, Macchi V, Ciarleglio FA, Palu G, De Caro R, Viel G, Favia G: Hyperparathyroidism-jaw tumor syndrome: a report of three large kindred. Langenbeck Arch Surg. 2009, 394: 817-825. 10.1007/s00423-009-0511-y.CrossRef

26.

Raue F, Haag C, Frank-Raue K: [Hyperparathyroidism-jaw tumor syndrome. A hereditary form of primary hyperparathyroidism with parathyroid carcinoma]. Deutsche medizinische Wochenschrift. 2007, 132: 1459-1462. 10.1055/s-2007-982052.PubMedCrossRef

27.

Moon SD, Park JH, Kim EM, Kim JH, Han JH, Yoo SJ, Yoon KH, Kang MI, Lee KW, Son HY, Kang SK, Oh SJ, Kim KM, Yoon SJ, Park JG, Kim IJ, Kang HC, Hong SW, Kim KR, Cha BY: A Novel IVS2-1G > A mutation causes aberrant splicing of the HRPT2 gene in a family with hyperparathyroidism-jaw tumor syndrome. J Clin Endocr Metab. 2005, 90: 878-883. 10.1210/jc.2004-0991.PubMedCrossRef

28.

Teh BT, Farnebo F, Kristoffersson U, Sundelin B, Cardinal J, Axelson R, Yap A, Epstein M, Heath H, Cameron D, Larsson C: Autosomal dominant primary hyperparathyroidism and jaw tumor syndrome associated with renal hamartomas and cystic kidney disease: linkage to 1q21-q32 and loss of the wild type allele in renal hamartomas. J Clin Endocr Metab. 1996, 81: 4204-4211.PubMed

29.

Mallette LE, Malini S, Rappaport MP, Kirkland JL: Familial cystic parathyroid adenomatosis. Ann Int Med. 1987, 107: 54-60. 10.7326/0003-4819-107-1-54.PubMedCrossRef

30.

Rekik N, Ben Naceur B, Mnif M, Mnif F, Mnif H, Boudawara T, Abid M: Hyperparathyroidism-jaw tumor syndrome: a case report. Annales d’endocrinologie. 2010, 71: 121-126. 10.1016/j.ando.2009.09.004.PubMedCrossRef

31.

Dinnen JS, Greenwoood RH, Jones JH, Walker DA, Williams ED: Parathyroid carcinoma in familial hyperparathyroidism. J Clin Pathol. 1977, 30: 966-975. 10.1136/jcp.30.10.966.PubMedPubMedCentralCrossRef

32.

Cavaco BM, Guerra L, Bradley KJ, Carvalho D, Harding B, Oliveira A, Santos MA, Sobrinho LG, Thakker RV, Leite V: Hyperparathyroidism-jaw tumor syndrome in Roma families from Portugal is due to a founder mutation of the HRPT2 gene. J Clin Endocr Metab. 2004, 89: 1747-1752. 10.1210/jc.2003-031016.PubMedCrossRef

33.

Schmidt BP, Bradrick JP, Gabali A: Hyperparathyroidism-jaw tumor syndrome: a case report. J Oral Maxillofac Surg. 2009, 67: 423-427. 10.1016/j.joms.2008.07.015.PubMedCrossRef

34.

Szabo J, Heath B, Hill VM, Jackson CE, Zarbo RJ, Mallette LE, Chew SL, Besser GM, Thakker RV, Huff V, Leppert MF, Heath H: Hereditary hyperparathyroidism-jaw tumor syndrome: the endocrine tumor gene HRPT2 maps to chromosome 1q21-q31. Am J Hum Genet. 1995, 56: 944-950.PubMedPubMedCentral

35.

Howell VM, Zori RT, Stalker HJ, Williams C, Jesse N, Nelson AE, Robinson BG, Marsh DJ: A molecular diagnosis of hyperparathyroidism—jaw tumor syndrome in an adolescent with recurrent kidney stones. J Pediatr. 2004, 145: 567-10.1016/j.jpeds.2004.04.023.PubMedCrossRef

36.

Warnakulasuriya S, Markwell BD, Williams DM: Familial hyperparathyroidism associated with cementifying fibromas of the jaws in two siblings. Oral Surg Oral Med Oral Pathol. 1985, 59: 269-274. 10.1016/0030-4220(85)90165-3.PubMedCrossRef

37.

Fujikawa M, Okamura K, Sato K, Mizokami T, Tamaki K, Yanagida T, Fujishima M: Familial isolated hyperparathyroidism due to multiple adenomas associated with ossifying jaw fibroma and multiple uterine adenomyomatous polyps. Eurn J Endocrinol. 1998, 138: 557-561. 10.1530/eje.0.1380557.CrossRef

38.

Bradley ES, Leake D: Ossifying fibroma involving the maxilla and mandible: report of a case. Oral Surg Oral Med Oral Pathol. 1968, 26: 605-614. 10.1016/0030-4220(68)90424-6.PubMedCrossRef

39.

Takeda Y, Fujioka Y: Multiple cemento-ossifying fibroma. Int J Oral Maxillofac Surg. 1987, 16: 368-371. 10.1016/S0901-5027(87)80161-3.PubMedCrossRef

40.

Hauser MS, Freije S, Payne RW, Timen S: Bilateral ossifying fibroma of the maxillary sinus. Oral Surg Oral Med Oral Pathol. 1989, 68: 759-763. 10.1016/0030-4220(89)90168-0.PubMedCrossRef

41.

Yih WY, Pederson GT, Bartley MH: Multiple familial ossifying fibromas: relationship to other osseous lesions of the jaws. Oral Surg Oral Med Oral Pathol. 1989, 68: 754-758. 10.1016/0030-4220(89)90167-9.PubMedCrossRef

42.

Khanna JN, Andrade NN: Giant ossifying fibroma. Case report on a bimaxillary presentation. Int J Oral Maxillofac Surg. 1992, 21: 233-235. 10.1016/S0901-5027(05)80226-7.PubMedCrossRef

43.

Hwang EH, Kim HW, Kim KD, Lee SR: Multiple cemento-ossifying fibroma: report of an 18-year follow-up. Dento Maxillo Facial Radiol. 2001, 30: 230-234. 10.1038/sj.dmfr.4600608.CrossRef

44.

Bertolini F, Caradonna L, Bianchi B, Sesenna E: Multiple ossifying fibromas of the jaws: a case report. J Oral Maxillofac Surg. 2002, 60: 225-229. 10.1053/joms.2002.29832.PubMedCrossRef

45.

Barberi A, Cappabianca S, Colella G: Bilateral cemento-ossifying fibroma of the maxillary sinus. Brit J Radiol. 2003, 76: 279-280.PubMedCrossRef

46.

Stergiou GC, Zwahlen RA, Gratz KW: [Multiple cemento-ossifying fibromas of the jaw: a very rare diagnosis]. Schweiz Monatsschr Zahnmed. 2007, 117: 236-244.PubMed

47.

Chindia ML, Dimba EA, Moshy J, Limo A, Otwoma JG, Guthua SW: Synchronous occurrence of ossifying fibroma of the mandible and maxilla: a case report. Dent Update. 2008, 35: 705-707.PubMed

48.

Ribeiro AC, Carlos R, Diaz KP, Gouvea AF, Vargas PA: Bilateral central ossifying fibroma affecting the mandible: report of an uncommon case and critical review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011, 111: e21-e26.PubMedCrossRef

49.

Akcam T, Altug HA, Karakoc O, Sencimen M, Ozkan A, Bayar GR, Gunhan O: Synchronous ossifying fibromas of the jaws: a review. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012, 114: S120-S125. 10.1016/j.oooo.2011.08.007.PubMedCrossRef

50.

Agarwal N, Gupta P, Gupta P, Naik S, Upadhyay N: Recurrent bimaxillary radiopacities: a rare case report. Contemp Clin Dent. 2012, 3: S103-S108.PubMedPubMedCentralCrossRef

51.

Alsharif MJ, Sun ZJ, Chen XM, Wang SP, Zhao YF: Benign fibro-osseous lesions of the jaws: a study of 127 Chinese patients and review of the literature. Int J Surg Pathol. 2009, 17: 122-134.PubMedCrossRef

52.

Idowu BD, Al-Adnani M, O’Donnell P, Yu L, Odell E, Diss T, Gale RE, Flanagan AM: A sensitive mutation-specific screening technique for GNAS1 mutations in cases of fibrous dysplasia: the first report of a codon 227 mutation in bone. Histopathology. 2007, 50: 691-704. 10.1111/j.1365-2559.2007.02676.x.PubMedCrossRef

53.

Tabareau-Delalande F, Collin C, Gomez-Brouchet A, Decouvelaere AV, Bouvier C, Larousserie F, Marie B, Delfour C, Aubert S, Rosset P, de Muret A, Pages JC, de Pinieux G: Diagnostic value of investigating GNAS mutations in fibro-osseous lesions: a retrospective study of 91 cases of fibrous dysplasia and 40 other fibro-osseous lesions. Modern Pathol. 2013, 26: 911-921. 10.1038/modpathol.2012.223.CrossRef

54.

Bradley KJ, Cavaco BM, Bowl MR, Harding B, Cranston T, Fratter C, Besser GM, Conceicao Pereira M, Davie MW, Dudley N, Leite V, Sadler GP, Seller A, Thakker RV: Parafibromin mutations in hereditary hyperparathyroidism syndromes and parathyroid tumours. Clin Endocrinol. 2006, 64: 299-306. 10.1111/j.1365-2265.2006.02460.x.CrossRef

55.

Sarquis MS, Silveira LG, Pimenta FJ, Dias EP, Teh BT, Friedman E, Gomez RS, Tavares GC, Eng C, De Marco L: Familial hyperparathyroidism: surgical outcome after 30 years of follow-up in three families with germline HRPT2 mutations. Surgery. 2008, 143: 630-640. 10.1016/j.surg.2007.12.012.PubMedCrossRef

56.

Guarnieri V, Bisceglia M, Bonfitto N, Cetani F, Marcocci C, Minisola S, Battista C, Chiodini I, Cole DE, Scillitani A: Re: Familial hyperparathyroidism: surgical outcome after 30 years of follow-up in three families with germline HRPT2 mutations. Surgery. 2008, 144: 839-840.PubMedCrossRef

57.

McCarthy EF: Fibro-osseous lesions of the maxillofacial bones. Head Neck Pathol. 2013, 7: 5-10. 10.1007/s12105-013-0430-7.PubMedPubMedCentralCrossRef

58.

Summerlin DJ, Tomich CE: Focal cemento-osseous dysplasia: a clinicopathologic study of 221 cases. Oral Surg Oral Med Oral Pathol. 1994, 78: 611-620. 10.1016/0030-4220(94)90174-0.PubMedCrossRef

59.

Su L, Weathers DR, Waldron CA: Distinguishing features of focal cemento-osseous dysplasia and cemento-ossifying fibromas: II. A clinical and radiologic spectrum of 316 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1997, 84: 540-549. 10.1016/S1079-2104(97)90271-7.PubMedCrossRef

60.

Gunduz K, Avsever H, Karacayli U, Senel B, Piskin B: Florid cemento-osseous dysplasia: a case report. Braz Dent J. 2009, 20: 347-350.PubMed

61.

Coleman H, Altini M, Kieser J, Nissenbaum M: Familial florid cemento-osseous dysplasia–a case report and review of the literature. J Dent Assoc S Afr. 1996, 51: 766-770.PubMed

62.

Toffanin A, Benetti R, Manconi R: Familial florid cemento-osseous dysplasia: a case report. J Oral Maxillofac Surg. 2000, 58: 1440-1446. 10.1053/joms.2000.16638.PubMedCrossRef

63.

Abdelsayed RA, Eversole LR, Singh BS, Scarbrough FE: Gigantiform cementoma: clinicopathologic presentation of 3 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001, 91: 438-444. 10.1067/moe.2001.113108.PubMedCrossRef

64.

Shah S, Huh KH, Yi WJ, Heo MS, Lee SS, Choi SC: Follow-up CT findings of recurrent familial gigantiform cementoma of a female child. Skelet Radiol. 2012, 41: 341-346. 10.1007/s00256-011-1245-9.CrossRef

65.

Capodiferro S, Maiorano E, Giardina C, Lacaita MG, Lo Muzio L, Favia G: Osteoblastoma of the mandible: clinicopathologic study of four cases and literature review. Head Neck. 2005, 27: 616-621. 10.1002/hed.20192.PubMedCrossRef

66.

Hammas N, Laila C, Youssef ALM, Hind EF, Harmouch T, Siham T, Afaf A: Can p63 serve as a biomarker for giant cell tumor of bone? A Moroccan experience. Diagn Pathol. 2012, 7: 130-10.1186/1746-1596-7-130.PubMedPubMedCentralCrossRef

67.

Jain D, Jain VK, Vasishta RK, Ranjan P, Kumar Y: Adamantinoma: a clinicopathological review and update. Diagn Pathol. 2008, 3: 8-10.1186/1746-1596-3-8.PubMedPubMedCentralCrossRef

68.

Armah HB, McGough RL, Goodman MA, Gollin SM, Surti U, Parwani AV, Rao U: Chondromyxoid fibroma of rib with a novel chromosomal translocation: a report of four additional cases at unusual sites. Diagn Pathol. 2007, 2: 44-10.1186/1746-1596-2-44.PubMedPubMedCentralCrossRef

Title: Two cases of multiple ossifying fibromas in the jaws
Authors: Ting-Ting Wang
Ran Zhang
Lin Wang
Yan Chen
Qing Dong
Tie-Jun Li
Publication date: 01-12-2014
Publisher: BioMed Central
Published in: Diagnostic Pathology / Issue 1/2014
Electronic ISSN: 1746-1596
DOI: https://doi.org/10.1186/1746-1596-9-75

At a glance: The ONWARDS insulin icodec trials

Springer Medicine

Two cases of multiple ossifying fibromas in the jaws

Abstract

Background

Methods

Results

Conclusions

Virtual slides

At a glance: The ONWARDS insulin icodec trials

Springer Medicine

Abstract

Background

Methods

Results

Conclusions

Virtual slides

Please log in to get access to this content

Other articles of this Issue 1/2014

Automated Selection of Hotspots (ASH): enhanced automated segmentation and adaptive step finding for Ki67 hotspot detection in adrenal cortical cancer

Radiological and pathological characteristics of giant cell tumor of bone treated with denosumab

High expression of microRNA-130b correlates with poor prognosis of patients with hepatocellular carcinoma

Occipital anaplastic oligodendroglioma with multiple organ metastases after a short clinical course: a case report and literature review

A time course-dependent metastatic gene expression signature predicts outcome in human metastatic melanomas

Expression of serum amyloid A in uterine cervical cancer