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

INTRODUCTION

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).

CASE DESCRIPTION

DISCUSSION

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).^1–6

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.^16–18

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

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.

CLINICAL SIGNIFICANCE

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).

ACKNOWLEDGMENTS

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.

ORCID

Dwi Juliana Dewi https://orcid.org/0000-0002-8670-066X

Respati Ranakusuma https://orcid.org/0000-0002-6678-7876

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