Juvenile ossifying fibroma (JOF) and its variants, including juvenile psammomatoid ossifying fibroma (JPOF), represent rare yet clinically significant benign fibro-osseous lesions that primarily occur in children and young adolescents. They can be found in diverse anatomical sites such as the jaw, nasal cavity, paranasal sinuses, and orbit. JOF exhibits an aggressive nature, necessitating early radiological detection and surgical intervention. Similarly, JPOF, with a locally malignant potential, requires surgical removal, typically conducted through endoscopic approaches. We report a case of a 5-year-old girl with JPOF arising in the ethmoid, revealed by recurrent epistaxis and proptosis. The text emphasizes the importance of early diagnosis through histopathology as a diagnostic tool and underscores the need for appropriate management.

OBJECTIVE: The purpose of this study is to evaluate the association between hyperparathyroidism (PHPT), parathyroid hormone levels, and calcium levels in patients diagnosed with benign fibro-osseous lesions such as fibrous dysplasia (FD), ossifying fibroma (OF), central giant cell granulomas (GCG).
METHODS: This is a retrospective, single-center study from a sample of patients who underwent surgical treatment of FD, OF, and GCG at Mayo Clinic between 1996 and 2021. Patient demographics, history of PHPT, histopathological diagnosis, and relevant laboratory values such as parathyroid hormone (PTH), serum calcium, vitamin D, and alkaline phosphatase were collected.
RESULTS: Of the patients diagnosed with FD (n = 64), OF (n = 24), and GCG (n = 5), a diagnosis of PHPT was found in 2 patients (3.1%), 1 patient (4.2%), and 0 patients (0%), respectively. Elevated PTH levels (>65 pg/mL) were observed in 3 patients (4.7%) with FD, 1 patient (4.2%) with OF, and 1 patient (20%) with GCG. Mean (standard deviation) calcium levels were 9.3 (0.6) mg/dL in the FD group, 9.4 (0.5) mg/dL in the OF group, and 9.3 (0.6) mg/dL in the GCG group. Patients with fibro-osseous jaw tumors including FD, OF, and GCG may have increased risk of PHPT compared to the general population.
CONCLUSIONS: Patients with benign jaw tumors including FD, OF, and GCG may have increased risk of PHPT compared to the general population. Surgeons treating these benign tumors need to be cognizant of these findings, obtain appropriate laboratory studies, and incorporate multidisciplinary care including endocrinologists, endocrine surgeons, and maxillofacial surgeons.

Background Fibro-osseous (FO) lesions are slow-growing benign lesions in the paranasal sinuses. They include osteomas, fibrous dysplasia (FD), and ossifying fibro-ma (OF). Fibro-osseous (FO) lesions are frequently asymptomatic, and they are incidentally found on imaging. They are characterized by different histological, radiological, and clinical variants. Depending on symptoms, size, location, and extension, the treatment strategy varies significantly for these lesions. Objective We aim to compare the age, onset, gender, clinical presentation, postoperative improvement, and complications of a fibro-osseous lesion in the paranasal sinuses. Methods A retrospective analysis was done targeting patients diagnosed with benign fibro-osseous (FO) lesions, and the incidence among 403 patients who underwent functional endoscopic sinus surgery (FESS) at Aseer Central Hospital, Kingdom of Saudi Arabia, was reviewed from January 2013 to January 2022. Results A total of seven patients were found; five patients were diagnosed with osteoma, and two were diagnosed with fibrous dysplasia. There were no ossifying fibroma cases. The patients\' mean age was 25.5 ± 12.9 years old. Four (57.1%) patients were males, and three (42.9%) were females, with a male/female ratio of 1.25:1. The most common locations were the frontal sinus and ethmoid sinus, and the two cases of fibrous dysplasia involved almost all facial bones. The endonasal endoscopic approach was chosen to treat all seven patients. Conclusions There are differences in the onset age, location, and complications postoperatively among osteoma and fibrous dysplasia patients. Osteoma most commonly occurs in the frontal sinus, while fibrous dysplasia involved all facial bones in our study. Endoscopic surgery is currently the primary strategy for treatment.

BACKGROUND: Fibro-osseous lesions (FOL) of the jaw represent a rare, benign group of lesions that share similar clinical, radiological, and histopathological features and are characterized by progressive, variable replacement of healthy bone tissue by fibrous connective tissue.
METHODS: This retrospective study aimed to evaluate the incidence of fibro-osseous lesions and to reassess the efficacy of case-specific treatment management from a clinical, radiological, and histopathological perspective based on 14 years of data.
RESULTS: Forty-four patients with a radiological and/or histopathological diagnosis of benign FOLs were identified and re-evaluated. Cemento-osseous dysplasia was the most common group of FOLs present in our patient cohort (45%), followed by ossifying fibroma (39%) and fibrous dysplasia (16%). The diagnostic imaging technique of choice was CBCT (68%), followed by PAN (18%), with most patients (95 %) additionally undergoing biopsy. The mean age of the patients at the time of diagnosis was 40.54 ± 13.7 years, with most lesions being located in the mandible (86%), with females being predominantly affected (73%).
CONCLUSIONS: An interdisciplinary approach that analyzes all case-specific factors, including demographic data, medical history, intraoperative findings, and, most importantly, histopathological and radiological features, is essential for an accurate diagnosis and key to avoiding inappropriate treatment.

BACKGROUND: Fibrous dysplasia (FD) and cemento-ossifying fibroma (COF) are the most common gnathic fibro-osseous lesions. These diseases exhibit remarkable overlap of several clinicopathological aspects, and differential diagnosis depends on the combination of histopathological, radiographic, and clinical aspects. Their molecular landscape remains poorly characterized, and herein, we assessed their proteomic and phosphoproteomic profiles.
METHODS: The quantitative differences in protein profile of FD and COF were assessed by proteomic and phosphoproteomic analyses of formalin-fixed paraffin-embedded tissue samples. Pathway enrichment analyses with differentially regulated proteins were performed.
RESULTS: FD and COF exhibited differential regulation of pathways related to extracellular matrix organization, cell adhesion, and platelet and erythrocytes activities. Additionally, these lesions demonstrated distinct abundance of proteins involved in osteoblastic differentiation and tumorigenesis and differential abundance of phosphorylation of Ser61 of Yes-associated protein 1 (YAP1).
CONCLUSIONS: In summary, despite the morphological similarity between these diseases, our results demonstrated that COF and DF present numerous quantitative differences in their proteomic profiles. These findings suggest that these fibro-osseous lesions trigger distinct molecular mechanisms during their pathogenesis. Moreover, some proteins identified in our analysis could serve as potential biomarkers for differential diagnosis of these diseases after further validation.

OBJECTIVE: Benign fibro-osseous lesions (BFOLs) are heterogeneous intraosseous disease processes sharing overlapping histopathologic features and demonstrate a wide range of biological behavior. The aim of this study is to highlight the importance of clinical and radiological findings for the definitive histopathologic diagnosis of benign fibro-osseous lesions of the jaws.
METHODS: This is a cross-sectional retrospective study evaluating the three main groups of BFOLs: Cemento-osseous dysplasia, ossifying fibromas, and fibrous dysplasia. Previously diagnosed BFOL cases were searched for thoroughly from the archives of Istanbul University, Institute of Oncology, Department of Tumor Pathology in between 2005 and 2015. Clinical and radiological data of these cases were identified and recorded. The histopathologic features were reevaluated from the H&E-stained slides.
RESULTS: A total of 276 BFOL cases were identified and reevaluated for the study. The most common BFOL type was cemento-osseous dysplasia (n = 135), followed by cemento-ossifying fibroma (n = 77), fibrous dysplasia (n = 53) and juvenile ossifying fibroma (n = 11). The female/male ratio was 3.4:1 with a strong predilection for the 4th decade (n = 102). The mandible (n = 209) was the predominantly affected jaw. Prominent osteoblastic rimming around the bony lesion was commonly observed for cemento-ossifying fibroma (n = 60/77), followed by cemento-osseous dysplasia (n = 23/135). Predominant ossification patterns showed some differences among the groups. The most common radiological feature was the mixed radiolucent/radiopaque internal structure.
CONCLUSIONS: Our results document various clinical, radiological and histopathologic findings of BFOLs. Even some histopathologic differences are observed, clinical and radiographic correlation is mandatory prior to the definitive histopathologic diagnosis of BFOLs for the prevention of possible misdiagnoses or inappropriate treatments.

To compare the clinical characteristics of osteoma, ossifying fibroma (OF) and fibrous dysplasia (FD) in the paranasal sinus and craniofacial regions.
Totally 282 patients (112 males, 170 females) with osteoma (161), OF (44), and FD (77) involving the paranasal sinus, skull base and orbit treated surgically from January 2012 to August 2018 were analyzed retrospectively.
For osteoma, OF and FD, the onset ages were 40.3 (6-75), 24.5 (5-62), and 15 (1-63) years, and the most common locations were ethmoid sinus (49.7%), maxilla (36.4%) and maxilla (49.4%), respectively. There were significant differences of the preoperative serum alkaline phosphatase (ALP) levels between patients with osteoma (65 [25,184] U/L), OF (85.5 [41,474] U/L), and FD (104 [39,362] U/L) (Z = 44.9, P < .05). The ALP levels of OF and FD patients were comparable between monostotic and polyostotic lesions (P > .05). The recurrent rates of osteoma, OF, and FD were 0%, 13.6%, and 15.6%, respectively. The recurrent OF cases had significantly higher ALP level than the primary ones (283.5 [108,474] U/L vs. 76 [41,348] U/L, U = 14, P < .05).
There are differences in the onset age, location, and recurrence rate among osteoma, OF and FD involving the paranasal sinus and craniofacial region. Osteoma most commonly occurs in the ethmoid sinus, while OF and FD involve the maxilla most and are more likely to involve the orbit and the skull base, respectively. Endoscopic surgery is currently the main method for treatment, but individualized treatment regimen should be developed for patients.
IV Laryngoscope, 131:E1-E7, 2021.

Considered as an aggressive counterpart of central ossifying fibroma (OF), juvenile ossifying fibroma (JOF) is a benign fibro-osseous neoplasm characterized by an unpredictable destructive behavior, elevated morbidity, mutilating treatment and high potential for local recurrences. The aim of this study is to compare the analysis for cell proliferation and vascular markers between JOF and OF. Cell proliferation index was measured by Ki-67 and Mcm-2 expression and microvessel density (MVD) was obtained by the immunoexpression of CD34/CD105. We observed a reduced expression of vascular markers, where MVD for CD34 was significantly higher in JOF than in OF (p = 0.009), but no statistical difference was found for CD105. JOF and OF showed low expression for Ki-67 and Mcm-2 and no difference was noted between both, suggesting that other mechanisms such as anti-apoptotic and/or pro-autophagic pathways or even increased expression of matrix metalloproteinases may be responsible for the aggressiveness of JOF.

BACKGROUND: Ossifying fibroma in the jaws is a benign tumor and easily recurs in children, of which the treatment methods and prognosis still remain controversial. In this study, we aimed to review the clinicopathological characteristics, treatment, and prognosis of ossifying fibroma in the jaws of children, and offer recommendations for clinical decision-making. METHODS: A retrospective study was carried out on patients below the age of 18 years with ossifying fibroma in the jaws. Patients with complete clinical, pathological, and radiological records were included and followed-up. RESULTS: Sixty-three cases were collected with a preliminary search. After screening, fifty patients were included for general information analysis, of which forty-two patients were included in the recurrence analysis. Twelve patients showed a relapse, with a recurrence rate of 28.6% (12/42). The recurrence rates in cases with different surgical approaches and different X-ray boundaries were statistically different. Besides, twenty-three patients underwent reconstruction by free tissue grafting and the success rate was 96% (22/23). CONCLUSIONS: There was significant difference in the recurrence rates among different X-ray manifestations and surgical methods. An extended resection and reconstruction with free tissue grafting was a reliable method with a 96% success rate.

BACKGROUND: Fibro-osseous lesions are a diverse group of bone disorders and include developmental, reactive or dysplastic diseases and neoplasms. They share overlapping clinical, radiographic and histopathologic features and demonstrate a wide range of biological behaviour.
OBJECTIVE: To evaluate the characteristics, treatment and outcome of benign fibro-osseous lesions of the jaws.
METHODS: All patients with fibro-osseous lesions of the jaws treated at the department of Oral and Maxillofacial Surgery of the Kamineni Institute of Dental Sciences from 2007 to 2013 were included in this study.
RESULTS: Six males and four females were treated. Juvenile ossifying fibroma was most often encountered (40 %), and the mandible was the most frequent location (70 %). Main clinical feature in most of the cases was a painless expansile swelling with facial asymmetry, and radiologically mixed (radiolucent and radiopaque lesions) were seen in majority of cases. All cases were surgically treated and histopathologically confirmed. Segmental ostectomy was performed in six cases; maxillectomy was done in one case and excision along with margin in three cases. Mean follow-up was of 3.3 years with no recurrence.
CONCLUSIONS: Fibro-osseous lesions, although sharing similar microscopic features, exhibit a variety of clinical behavior rendering their treatment highly individualized. Radical treatment is the choice to achieve an outcome without recurrence.