Otorhinolaryngology Clinics: An International Journal
Volume 15 | Issue 3 | Year 2023

Type of Surgery to Decrease Postoperative Recurrence in Ossifying Fibroma: Case Report

Dwi Juliana Dewi1, Marlinda Adham2, Respati Ranakusuma3

1,2Department of Otorhinolaryngology Head and Neck Surgery, Universitas Indonesia, Daerah Khusus Ibukota, Jakarta, Indonesia

3The Center for Clinical Epidemiology and Evidence-Based Medicine (CEEBM), Universitas Indonesia, Daerah Khusus Ibukota, Jakarta, Indonesia

Corresponding Author: Dwi Juliana Dewi, Department of Otorhinolaryngology Head and Neck Surgery, Universitas Indonesia, Daerah Khusus Ibukota, Jakarta, Indonesia, e-mail:

How to cite this article: Dewi DJ, Adham M, Ranakusuma R. Type of Surgery to Decrease Postoperative Recurrence in Ossifying Fibroma: Case Report. Int J Otorhinolaryngol Clin 2023;15(3):153–157.

Source of support: Nil

Conflict of interest: None

Patient consent statement: The author(s) have obtained written informed consent from the patient’s parents/legal guardians for publication of the case report details and related images.

Received on: 07 July 2022; Accepted on: 28 May 2023; Published on: 09 January 2024


Background: Ossifying fibroma (OF) in craniofacial is a rare disease, benign, locally aggressive fibro-osseous tumor. In the recent 2017 WHO classifications, OF was divided into two types, ossifying fibroma of odontogenic origin and juvenile ossifying fibroma (JOF). Determining the right surgical treatment to reduce the postoperative recurrence rate is incredibly challenging.

Case description: The author reports two cases of OF in paranasal sinuses with disease onset progressed from prepubertal age. The first case is an example of a recurrent case after undergoing conservative surgery and the second case is a new case. All cases underwent radical surgery with subtotal maxillectomy and reconstructive surgery in one stage. After observing all patients until one year, there were no signs of recurrence through clinical and endoscopic examination.

Discussion: There are 2 types of surgery that are compared in this case report, conservative surgery procedure, and radical surgery. Conservative surgical procedures include curettage, enucleation, or peripheral osteotomies. Several studies showed high recurrence levels when curettage or enucleation is conducted in OF patients, residue caused by incomplete excision is most the common reason that is easily caused by conservative surgery. Radical surgery such as open maxillectomy shows as a promising approach for degrading the level of recurrence.

Conclusion: Juvenile ossifying fibroma, especially trabecular juvenile ossifying fibroma (TrJOF), show a high recurrence percentage compared to other type. The first-choice management for treating OF was the surgical approach. Types of surgery choose to depend on the aggressiveness and morbidity of the disease. Radical surgery was proven better to decrease the level of recurrence compared with conservative surgery.

Keywords: Case report, Chondroblastoma, Curettage, Fibro-osseous lesion, Maxilla, Maxillectomy, Ossifying fibroma, Osteotomy.


Fibro-osseous lesion is defined as benign a tumor with fibrous cellular stroma which comprises focus mineralization or ossification in normal bone.1 Applani MC described them as “replacing normal bone in the jaw by fibro-osseous tissue sometimes followed with calcification”. The lesion frequently arises in the craniofacial region, in the jaw, nasal, paranasal sinuses, and orbital. Divide into three types, there are osteomas, fibrous dysplasia (FD), and ossifying fibroma (OF). Osteomas is a prevalence type compared with others.1,2

Ossifying fibroma often develops in the head-neck region. The lesion reported the most prevalence in young adulthood and dominated in females. In 2017, World Health Organization (WHO) was reclassified with cemento-ossifying fibroma which is part of the odontogenic tumor, and the other type was juvenile ossifying fibroma (JOF). Juvenile ossifying fibroma is divided into two types, psammomatoid juvenile ossifying fibroma (PsJOF) and trabecular juvenile ossifying fibroma (TrJOF). Psammomatoid juvenile ossifying fibroma is detected in young adults compared with TrJOF prevalence in the younger age groups. Psammomatoid juvenile ossifying fibroma is predominantly located in the paranasal sinus and orbital region, whereas TrJOF is mostly located in the jaws.3,4

The treatment of JOF varies by the subtype and aggressiveness of the tumor. Conservative surgery is indicated in some cases, and radical surgery including a total resection of tumor may be needed in selective cases. This case report describes two clinical cases of OF and finds the surgical treatment to reduce postoperative recurrence in OF based on searching the evidence-based journal (Fig. 1).

Figs 1A and B: (A) The paranasal sinus CT-scan with contrast of the 1st patient revealed a mass extended from the left frontal region to the left sphenoid wing, expansion to the glabella, left sphenoid, and ethmoid sinus with signs of calcification, hyperostosis, and bone destruction. The left orbit was pushed inferiorly; (B) Histology result was proliferative cell, spindle and stellate form, core of cell was uniform, with smooth chromatin. There was a sign of mineralized collagenous ossicles. No visible signs of malignancy


Case 1

A 20-year-old female patient has had a chief complaint of painless progressive mass in the superior region of the left orbital in the last 2 years. The preceding case history disclosed that the patient had undergone enucleation of mass and reconstruction surgery 5 years ago at Cipto Mangunkusumo National Hospital. The postoperative histopathology result was JOF. After 2 years, the patient developed swelling in the same region, which was rapidly increasing in size. The patient did not feel any change in vision. Elicited medical and family history did not reveal significant findings.

We performed subtotal maxillectomy and reconstruction in one stage. Reconstruction surgery was done by inserting a plate and screw at the infraorbital wall. Following the first week after the surgery, a healed facial incision was noted. Thereafter, the patient planned regular follow-up every 3 months.

Case 2

A 12-year-old boy patient visited our institution with a chief complaint painless progressive mass in the left eye in the last 1 year. There was no history of bleeding from the nose, blocked nose, or runny nose. The patient did not feel any change in vision. Exact medical and family history did not reveal significant findings.

We performed subtotal maxillectomy and reconstruction in one stage. Reconstruction surgery was made with titanium mesh and rib cartilage. Following the first week after the surgery, healed facial wound was noticed, and there was no sign of secondary infection. Thereafter, the patient planned regular follow up every 3 months.


Ossifying fibromas were first reported by Montgomery in 1927, as a benign fibro-osseous lesion. Fibrous dysplasia and OF are mostly known as similar entities, but different from each other. Generally, OF had a well-defined margin, different from FD, it is typically surrounded by a fibrous capsule that constructs an ill-defined margin. Ossifying fibroma may manifest as a sclerotic rim on plain radiographs and CT. In 1952, it was introduced that JOF as an aggressive form of ossifying fibroma predominantly in the craniofacial region of children.5 Even though study related to OF were reported for many years, the evidence about how to manage this disease still varies because lack of cases. It was seen from searching results that most types of studies related to OF were case reports, case series, or retrospective study (Fig. 2).16

Figs 2A and B: (A) Presurgery photograph. Sign of deformity in the left eye with lagophthalmos and proptosis; (B) Postsurgery photograph

Sex predominance in OF cases is more frequently diagnosed in males compared to females. Macdonald-Jankowski et al.7 reported that OF affected females more frequently than males with male to female ratio of 1:2.5. Site of lesion also reported similar predilection in each journal. Frequent predilection was mandible and another predilection site was in the maxilla, it is also found in the paranasal sinus despite lower intensity. 2,8,9

There are 2 types of surgery that are described in this report, conservative surgical procedures, and radical surgery. Conservative surgical procedure includes curettage, enucleation, or peripheral osteotomies. Several studies showed high recurrence levels when curettage or enucleation was conducted in OF patients. Adham and Dewi10 and Chrcanovic and Gomez6 reported conservative surgery proved more frequent postoperative recurrence. MacDonald-Jankowsky et al.7 delineated similar, conservative surgery such as curettage had the higher possibility to relapse after surgery. Several study reported that the complete removal of an OF lesion with safe margin when operated would decrease levels of recurrence. Similar studies reported that high recurrence level in OF are associated with incomplete excision of previous surgery. The residue caused by incomplete excision is most the common reason that is easily caused by conservative surgery.11,12

Radical surgery shows a more promising approach for degrading the level of recurrence. In systematic review conducted by Chrcanovic and Gomez6 shows zero recurrence when segmental resection was conducted in all types of JOF (TrJOF and PsJOF) and all predilection sites, followed by marginal resection also had zero recurrence percentage in all types of JOF and all predilection site but not statistically significance. Adham and Dewi10 in their systematic review reported postoperative recurrence following radical surgery was 10.6% compared with conservative surgery was 19.7%. A retrospective case series study which analyze in this conducted by Liu Ying et al.9 reported recurrence levels were 55.6% for local resection, and 3.8% for radical resection. Another study reported by Han et al.13 in their study from 6 patients who took local radical surgery had no evidence of recurrence. Suarez-Soto et al.14 and Kumar et al.2 also reported similar, in their studies describing recurrence radical surgery in several variants of ossifying fibroma showed zero percentage of recurrence. Liu Y et al.15 in their study reported from 7 patients who underwent radical surgery, the recurrence rate was 14.3% compared with conservative surgery was 50%. They also stated complete resection of the tumour with a radical approach may needed to lower the level of recurrence. Although slightly different in percentage value, the result of these studies is in line when compared. Another factor that may reduce the recurrence level was well-defined margin of tumor based on CT imaging. Chrcanovic and Gomez6 and Liu Ying et al.9 stated disparity between well-defined margin and ill-defined margin of CT imaging is almost 25%. If we look at the subtype of ossifying fibroma, TrJOF had a recurrence percentage higher than PsJOF and OF, this is in line with the characteristic of TrJOF that mention in several studies as an aggressive disease with high reccurence.10,11 Based on this, locally aggressive tumours with ill-defined margins on imaging consider a more aggressive treatment like radical surgery. Hence, doubts about the problem of defects that occur during the growth period, especially in pediatric patients could be managed by free flap.1618

Studies conducted by Chrcanovic and Gomez6 and Han J et al.13 reported an analogous average time of recurrence. It may recur between 1 and 2 years after surgery. The aggressive growth and tendency to reoccur is age-related and is seen more constantly in the youngish age group. After analyzing the relationship between them, Chrcanovic reported no association between age and recurrence rate when observed in statistical significance.6

Another type of surgery which did not discuss in this systematic review was endoscopic resection. Wang et al.3 reported a patient with OF who underwent endoscopic surgery, 13% of them developed recurrence. Juvenile ossifying fibroma is noted as a pathology of the recurrent cases that possibly behave more aggressively. Tumor extension is a contributing factor for selecting the surgical treatment. Most recurrent cases comprised site minimal in two paranasal sinuses, extended to the nasal cavity, orbital, and anterior cranial fossa. The endoscopic approach fails to manage tumor with advanced extension (more than 2 paranasal sinuses, orbital and cranial extension. Resection by endoscopic approach will minimize the site of surgery than make limitations for totally removing the mass.3

Adjuvant radiation therapy after surgery was reported to fail to decrease recurrence. Also considering the side effects on patients, uses after surgery should be restricted. Another study reported contraindications of radiotherapy in patients with OF due to an increased risk of malignant transformation from 0.4 to 40% (Fig. 3).19

Figs 3A and B: (A) The paranasal sinus CT-scan with the contrast of the 2nd patient revealed a mass extended from the left nasal bone, left paranasal sinus, left nasal cavity, nasal septum, and pushing the medial wall of the left eye causing non-axial proptosis in the left eye with the size of the mass was 4.1 cm × 3.7 cm × 4.5 cm with regular surfaces and strict lines border; (B) The pathognomonic sign of blue circle or psammoma bodies mostly found in psammomatoid JOF (PsJOF)

Radical surgery was shown as a promising approach to lowering the level of recurrence rather than simple curettage or enucleation. Adham and Dewi10 reported radical surgery can decrease postoperative recurrence in OF approximately 10.6%. However, the journals referred to in this study are in the form of systematic reviews of case series as well as primary studies in the form of case series which certainly have a low level of evidence (level 4). There was a lack of study which related to comparing types of surgery in OF. The type of this research is mostly with primary study in the form of case reports or case series. With this, it can be concluded that there is a knowledge gap in analyzing the OF cases. In the future, need for appropriate studies to assess the influence or prognostic factors related to postoperative recurrence in OF cases.


The first case was an example of a recurrence case after conservative surgery with mass enucleation. Pathology anatomy results revealed a TrJOF type, according to Adham and Dewi11 and Chrcanovic and Gomez.6 Trabecular juvenile ossifying fibroma subtype reported as an aggressive disease with high recurrence after surgery.

All patients underwent radical surgery with subtotal maxillectomy, the patients planned regular follow-up every 3 months, and there was no sign of recurrence after following observation 1 year after surgery. The patient was satisfied with the postoperative result. This condition is in accordance with evidence that concludes radical surgery was the promising therapy to manage postoperative recurrence in ossifying fibroma (Fig. 4).

Figs 4A to C: (A and B) Presurgery photograph. Sign of proptosis in left eye, no sign of lagophthalmos; (C) Postsurgery photograph


The author wishes to express her gratitude to all tutors who participated in the completion of this project. Especially, Marlinda Adham MD and Respati Ranakusuma MD provided their valuable suggestions, and their constructive guidance proved indispensable in completing this project.


Dwi Juliana Dewi

Respati Ranakusuma


1. Appiani MC, Verillaud B, Bresson D, et al. Ossifying fibromas of the paranasal sinuses: Diagnosis and management. Acta Otorhinolaryngol Ital 2015;35(5):355–361. DOI: 10.14639/0392-100X-533.

2. Kumar KAJ, Kishore PK, Mohan AP, et al. Management and treatment outcomes of maxillofacial fibro-osseous lesions: A retrospective study. J Maxillofac Oral Surg 2015;14(3):728–734. DOI: 10.1007/s12663-014-0726-5.

3. Wang H, Sun X, Liu Q, et al. Endoscopic resection of sinonasal ossifying fibroma: 31 cases report at an institution. Eur Arch Otorhinolaryngol 2014;271(11):2975–2982. DOI: 10.1007/s00405-014-2972-z.

4. Sarode SC, Sarode GS, Ingale Y, et al. Recurrent juvenile psammomatoid ossifying fibroma with secondary aneurysmal bone cyst of the maxilla: A case report and review of literature. Clin Pract 2018;8(3):1085. DOI: 10.4081/cp.2018.1085.

5. Maria A, Sharma Y, Malik M. Juvenile ossifying fibroma of mandible: A case report. J Maxillofac Oral Surg 2013;12(4):447–450. DOI: 10.1007/s12663-010-0122-8.

6. Chrcanovic BR, Gomez RS. Juvenile ossifying fibroma of the jaws and paranasal sinuses: A systematic review of the cases reported in the literature. Int J Oral Maxillofac Surg 2020;49(1):28–37. DOI: 10.1016/j.ijom.2019.06.029.

7. MacDonald-Jankowski DS. Ossifying fibroma: A systematic review. Dentomaxillofac Radiol 2009;38(8):495–513. DOI: 10.1259/dmfr/70933621.

8. Mohanty S, Gupta S, Kumar P, et al. Retrospective analysis of ossifying fibroma of jaw bones over a period of 10 years with literature review. J Maxillofac Oral Surg 2014;13(4):560–567. DOI: 10.1007/s12663-013-0545-0.

9. Liu Y, Shan X, Guo X, et al. Clinicopathological characteristics and prognosis of ossifying fibroma in the jaws of children: A retrospective study. Journal of Cancer 2018;8(17):3592–3597. DOI: 10.7150/jca.21556.

10. Adham M, Dewi JD. Comparison radical surgery versus conservative surgery to decrease post-operative recurrence in ossifying fibroma: Systematic review. J Oral Med Oral Surg 2020;26(44):1–9. DOI:

11. El-Mofty SK. Fibro-osseous lesions of the craniofacial skeleton: An update. Head Neck Pathol 2014;8(4):432–444. DOI: 10.1007/s12105-014-0590-0.

12. Bagheri SC. Oral and maxillofacial. In: Bagheri SC (Ed). Clinical review of oral and maxillofacial surgery. Atlanta: Mosby; 2008. p. 1–19.

13. Han J, Hu L, Zhang C, et al. Juvenile ossifying fibroma of the jaw: A retrospective study of 15 cases. Int J Oral Maxillofac Surg 2016; 45(3):368–376. DOI: 10.1016/j.ijom.2015.12.004.

14. Suarez-Soto A, Hermosa M, Minguez-Martínez I, et al. Management of fibro-osseous lesions of the craniofacial area presentation of 19 cases and review of the literature. Med Oral Patol Oral Cir Bucal 2013;18(3):479–485. DOI: 10.4317/medoral.18289.

15. Liu Y, Wang H, You M, et al. Ossifying fibromas of the jawbone: 20 cases. Dentomaxillofac Radiol 2010;39(1):57–63. DOI: 10.1259/dmfr/96330046.

16. Genden EM, Buchbinder D, Chaplin JM, et al. Reconstruction of the pediatric maxilla and mandible. Arch Otolaryngol Head Neck Surg 2000;126(3):293–300. DOI: 10.1001/archotol.126.3.293.

17. Zhang WB, Liang T, Peng X. Mandibular growth after paediatric mandibular reconstruction with the vascularized free fibula flap: A systematic review. Int J Oral Maxillofac Surg 2016;45(4):440–447. DOI: 10.1016/j.ijom.2015.12.014.

18. Güzel MZ, Arslan H, Saraç M. Mandibular condyle reconstruction with inlay application of autogenous costochondral graft after condylectomy: Cerrahpaşa’s technique. J Oral Maxillofac Surg 2007;65(4):615–620. DOI: 10.1016/j.joms.2005.12.061.

19. Liu JJ, Thompson LDR, Janisiewicz AM, et al. Ossifying fibroma of the maxilla and sinonasal tract: Case series. Allergy Rhinol 2007;8(1):e32–e36. DOI: 10.2500/ar.2017.8.0190.

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