Juvenile ossifying fibroma in a 24-year-old female with systemic comorbidities: a rare case report

Abstract

Introduction:

Juvenile ossifying fibroma (JOF) is a rare, benign, fibro-osseous lesion characterized by aggressive growth and potential for recurrence. It primarily affects children and adolescents, with two histopathological subtypes: trabecular JOF (TrJOF) and psammomatoid JOF (PsJOF). TrJOF typically presents in the jaws of younger individuals, often before the age of 15.

Case presentation:

A 24-year-old female with chronic kidney disease (CKD) and hypertension presented with progressive teeth displacement, mobility, and facial asymmetry. Radiographic evaluation revealed a well-defined radiolucent lesion affecting both jaws, displacing teeth, and perforating the cortical bone. Biopsy confirmed the diagnosis of TrJOF. Due to the aggressive growth and contraindications for chemotherapy or radiation therapy, surgical excision was performed. Postoperatively, the patient remains under observation, with ongoing management of CKD to prevent complications and ensure recovery.

Discussion:

TrJOF is an uncommon and aggressive variant of JOF that typically affects younger patients. This case is atypical due to the patient’s age and the involvement of both jaws. While JOF usually grows slowly, the lesions in this patient expanded rapidly, emphasizing the importance of prompt diagnosis and intervention. Surgical management remains the preferred approach, especially in cases complicated by systemic conditions like CKD. Recurrence rates vary widely, necessitating long-term follow-up.

Conclusion:

This case underscores the significance of early detection and multidisciplinary care in managing rare, aggressive jaw lesions such as TrJOF, particularly in patients with systemic comorbidities. Tailored surgical interventions and vigilant postoperative monitoring are crucial to achieving optimal outcomes and preventing recurrence.

Introduction

Fibro-osseous jaw lesions are an uncommon group of benign conditions marked by the gradual and varying replacement of normal bone tissue with fibrous connective tissue. These lesions share overlapping clinical, radiological, and histopathological characteristics^[1].

There are two distinct forms of benign fibro-osseous lesions of the jaws: conventional and juvenile ossifying fibroma (JOF). Based on the age of the patients, the most frequent locations of involvement, and the clinical behavior^[2]. The conventional form predominantly appears in the mandible of females aged 30–40 years. Conversely, the juvenile form, which is active and rare, tends to grow rapidly, typically affecting children or young adults and more frequently observed in the maxilla, and generally occurs before the age of 15^[1].

HIGHLIGHTS

• Rapid lesion growth and cortical perforation highlight TrJOF’s aggressive nature, necessitating early intervention.

• Uncommon 24-year-old female with bilateral JOF in both jaws, deviating from the typical age range.

JOF has two histopathological subtypes: trabecular JOF (TrJOF) and psammomatoid JOF (PsJOF). A distinguishing clinical feature between the two is their site of occurrence—PsJOF is typically found in the paranasal sinuses, while TrJOF predominantly affects the maxilla^[2].

Juvenile trabecular ossifying fibromas (JTOF) typically present as rapidly growing masses in individuals aged 5 to 15 years. Radiologically, they are well-defined and exhibit histological features consistent with ossifying fibroma^[3]. We report a case of a 24-year-old patient diagnosed with JOF involving both jaws, presenting atypically with rapid lesion expansion, accompanied by hypertension (HTN) and chronic kidney disease (CKD). This work has been reported in line with the SCARE 2025 criteria^[4].

Case presentation

We report a case of a 24-year-old female with CKD presenting with progressive displacement and mobility of teeth in the upper left premolar region (Fig. 1). Clinical examination revealed intact teeth with positive vitality tests and no gingival inflammation (Fig. 2), but notable facial asymmetry. Panoramic Radiograph demonstrated a well-defined radiolucent lesion displacing teeth and perforating the buccal cortical plate in the left maxilla, as well as a lingual lesion extending to the lower anterior teeth and premolars.

Figure 1.

Initial presentation showing displacement and mobility of teeth.

Figure 2.

Clinical examination showing intact teeth without gingival inflammation.

During the surgical procedure (Fig. 3), an incision was made in the gums starting from the area of the left lateral superior incisor and extending to the site of the unerupted second molar, concluding with a relaxing incision. A rounded tumor associated with the permanent premolar, which was also removed, was excised. Peripheral osteotomy was performed to reduce the likelihood of the lesion returning.

Figure 3.

Surgical incision and tumor excision path.

An intraosseous lesion associated with the crown and the root of an unerupted premolar, as well as with the roots of deciduous molar teeth, was excised and sent for histopathological assessment (Fig. 4). The histopathological examination confirmed a diagnosis of JTOF. Samples were preserved in formaldehyde and referred to the oral pathology department for further analysis (Fig. 5).

Figure 4.

Intraosseous lesion sent for histopathological analysis.

Figure 5.

Samples preserved in formaldehyde for biopsy.

Hematoxylin-eosin staining of the upper jaw lesion (Fig. 6) revealed thick irregular collagenous rims, cellular proliferation of spindle cells, and the presence of osteoclast giant cells with noticeable mitotic figures. Similarly, the lower jaw lesion biopsy demonstrated irregular interconnecting trabeculae of immature bone tissue containing coarse lacunae with plump osteocytes. The connective tissue was highly cellular, consisting of numerous proliferating spindle-shaped fibroblasts and multinucleated giant cells in some areas.

Figure 6.

Histopathological findings confirming diagnosis of trabecular juvenile ossifying fibroma.

Given the rapid growth of the lesions and contraindications for chemotherapy and radiation therapy due to the patient’s CKD, surgical excision was performed. Postoperatively, the patient is under observation with ongoing management of kidney failure to ensure optimal recovery and prevent recurrence.

Discussion

According to the World Health Organization, JOF is defined as a lesion affecting individuals under the age of 15, there are two histopathological variants of this condition: psammamatoid (PsJOF) and trabecular (TrJOF), which exhibit significant differences in their clinical and histopathological characteristics. The trabecular variant tends to occur at a younger average age (8 to 12 years) compared to the psammamatoid variant (16 to 33 years)^[5]. Moreover, Psammamatoid juvenile ossifying fibroma (PsJOF) is a rare form found in the extra gnathic craniofacial bones, particularly in the periorbital, frontal, and ethmoid regions^[6]. In contrast, TrJOF primarily affects the jaws, and there is ongoing debate regarding which jaw is more commonly involved^[7]. In our case, the 24-year-old patient has JOF noticed in both jaws accompanied by HTN and CKD. The majority of benign fibro-osseous lesions in the jaws are typically asymptomatic and exhibit slow growth. In certain instances, atypical clinical symptoms may arise in these lesions including facial asymmetry, teeth displacement, and movement^[8], which aligns with the observations in our case report. JOF is a type of fibro-osseous lesion found in the facial bones; unlike other lesions, it is classified as invasive fibro-osseous due to its aggressive nature and propensity for recurrence. JOF is considered a relatively uncommon fibro-osseous lesion of the jaws, predominantly affecting individuals younger than 15 years but this was an exception for our case^[9,10]. A higher preference exists for either the maxilla or mandible, with varying perspectives noted in the literature^[11]. The mandibular molar region is more frequently impacted than the maxilla. Both odontogenic fibromas (OF) and JOF show a nearly equal gender distribution in the general population. However, some studies report a slight male predominance for JOF^[11]. Our case, involving a young female, presents an exception to this pattern.

The posterior segments of the jaws are predominantly involved in most cases, aligning with earlier reports highlighting posterior mandible involvement^[6]. In our study, the lesions were arising within both jaws in the area of the upper left premolars and perforating the buccal cortical plate in the left maxillary area, along with a lingual lesion affecting the lower anterior teeth and premolars. Commonly, due to their slow and asymptomatic growth, these lesions can become significantly large upon initial diagnosis; nevertheless, some rare lesions may exhibit rapid enlargement, particularly in younger patients like ours. The recurrence rate for OF is notably low, whereas the recurrence rate for JOFs can vary, with reports indicating rates as high as 30% to 56%^[8]. This variability may be attributed to incomplete tumor excision. Smaller tumors can often be effectively managed through enucleation and curettage, while most experts recommend resection with 5-mm margins for larger, more aggressive tumors. In instances of recurrence, radical resection should be contemplated^[12]. The case presented here is atypical as it is associated with TrJOF, and there have been no documented instances of malignant transformation or inflammation of these lesions. Given the contraindications for chemotherapy and radiation therapy, surgical intervention was required to manage the quick expansion of lesions in both jaws and enhance the patient’s quality of life.

Conclusion

This case report highlights a rare and atypical presentation of JOF in a young adult patient with systemic comorbidities, including HTN and CKD. The involvement of both jaws with rapid lesion expansion underlines the importance of considering JOF in differential diagnoses beyond the typical pediatric population. Surgical excision remains the primary and most effective treatment modality, especially when other therapeutic options are contraindicated. This case emphasizes the need for early diagnosis, careful surgical planning, and multidisciplinary management to optimize patient outcomes and minimize recurrence risk.

Ethical approval

Our institution does not require ethical approval for reporting individual cases or case series.

Consent

Written informed consent was obtained from the patient’s parents/legal guardian for publication and any accompanying images. A copy of the written consent form is available for review by the editor-in-chief of this journal upon request.

Sources of funding

Not applicable.

Author contributions

A.H., M.A. contributed to drafting, reviewing, editing, and approving the final manuscript. A.A. contributed to drafting, editing, and approving the final manuscript. M.A. supervision.

Conflicts of interest disclosure

Not applicable.

Guarantor

Mohammad Obada Alsadi.

Research registration unique identifying number (UIN)

Not applicable.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Data availability statement

Data sharing is not applicable to this article.