Objective
A systematic review was undertaken to examine the spontaneous of craniofacial fibrous
dysplasia (FD) into osteosarcoma (OS).
Study Design
Five databases were searched, and data were evaluated in 2 subsets: (1) frequency
analysis involving only cross-sectional studies that specifically examined patients
for osteosarcomatous transformation, thereby allowing a calculation of the frequency
of transformation of craniofacial FD, and (2) case analysis of case reports and case
series of FD that underwent osteosarcomatous transformation. Quality assessment of
the studies in the frequency and case analyses was performed.
Results
Seven cross-sectional studies involving 779 patients in the frequency analysis documented
transformation of craniofacial FD into OS in 13 patients, for a frequency of 1.67%.
Of the 18 separate cases of osteosarcomatous transformation in the case analysis,
11 (61.1%) occurred in females, 11 (61.1%) developed from monostotic FD, and 7 (38.9%)
arose in the mandible, with 5 in the maxilla (27.8%) and 6 (33.3%) in other craniofacial
sites. The mean age of the patients at the time of malignant diagnosis was 36.4 years,
with an average of 11.3 years from FD diagnosis to the appearance of OS. The quality
of the articles ranged from fair to excellent.
Conclusions
The risk of osteosarcomatous transformation of craniofacial FD is low, but the possibility
of transformation should be taken into consideration by clinicians, as this will guide
treatment and follow-up regimens.
Abbreviations:
FD (Fibrous Dysplasia), OS (Osteosarcoma)To read this article in full you will need to make a payment
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References
- Maxillofacial fibrous dysplasia: a diagnostic challenge.BMJ Case Reports. 2016; 2016bcr2016215874
- Radiographic Diagnosis of Fibrous Dysplasia in Maxilla.Cureus. 2018; 10: e3127
- New knowledge of fibro-osseous lesions of bone.Int J Surg Pathol. 2010; 18: 62s-65s
- Maxillofacial fibro-osseous lesions.Current Diagnostic Pathology. 2006; 12: 1-10
- Fibrous dysplasia. Pathophysiology, evaluation, and treatment.J Bone Joint Surg Am. 2005; 87: 1848-1864
- Best practice management guidelines for fibrous dysplasia/McCune-Albright syndrome: a consensus statement from the FD/MAS international consortium.Orphanet J Rare Dis. 2019; 14: 139
- Osteosarcoma in fibrous dysplasia.Skeletal Radiol. 1988; 17: 163-170
- The PRISMA 2020 statement: an updated guideline for reporting systematic reviews.BMJ. 2021; 372: n71
DistillerSR V Distiller SR Inc.; 2022. https://www.evidencepartners.com. Accessed October 2020-May 2022.
- Spontaneous osteosarcoma in craniomaxillofacial fibrous dysplasia: clinical and computed tomographic features in 8 cases.Oral Surg Oral Med Oral Pathol Oral Radiol. 2014; 118: e24-e31
- Craniomaxillofacial fibrous dysplasia: a 10-year database 1996-2006.Br J Oral Maxillofac Surg. 2009; 47: 302-305
- Aggressive fibrous dysplasia of the maxillary sinus.Skeletal Radiol. 1993; 22: 563-568
- Fibrous Dysplasia: Clinicopathologic Presentation of 36 Cases.Turk Patoloji Derg. 2018; 34: 234-241
- Management of fibrous dysplasia. A report on 36 cases.Acta Orthop Belg. 2007; 73: 96-101
- Spontaneous malignant transformation in craniomaxillofacial fibrous dysplasia.J Craniofac Surg. 2013; 24: 141-145
- Craniofacial fibrous dysplasia: A 10-case series.Eur Ann Otorhinolaryngol Head Neck Dis. 2017; 134: 229-235
- Malignant transformation of fibrous dysplasia into chondroblastic osteosarcoma.Skeletal Radiol. 2002; 31: 103-106
- Malignant sarcomatous transformation of fibrous dysplasia.Head Neck Pathol. 2015; 9: 100-103
- Malignant transformation of monostotic fibrous dysplasia in the mandible.J Craniofac Surg. 2010; 21: 601-603
- Spontaneous conversion of fibrous dysplasia into osteosarcoma.J Craniofac Surg. 2011; 22: 959-961
- Sarcomatous transformation in the McCune-Albright syndrome.Oral Maxillofac Surg. 2012; 16: 217-220
- Spontaneous osteosarcoma transformation of fibrous dysplasia in maxilla.J Dent Sci. 2019; 14: 219-222
- Osteosarcoma of the Mandible Arising in Fibrous Dysplasia-A Case Report.J Oral Maxillofac Surg. 2016; 74 (2229.e1-29.e4)
- Osteosarcoma in a pregnant patient with McCune-Albright syndrome.Bone. 2009; 45: 603-608
- Advanced osteosarcoma of the maxilla: a case report.Med Oral Patol Oral Cir Bucal. 2008; 13: E492-E495
- A rare spontaneous osteosarcoma of the calvarium in a patient with long-standing fibrous dysplasia: CT and MR findings.Br J Radiol. 2008; 81: e31-e34
- Malignant transformation in fibrous dysplasia. A report of 3 cases.S Afr J Surg. 1990; 28: 80-82
- Albright's syndrome with reactivation of fibrous dysplasia secondary to pituitary adenoma and further complicated by osteogenic sarcoma. Report of a case.Oral Surg Oral Med Oral Pathol. 1984; 57: 616-619
- Fibrous dysplasia of the skull with acromegaly and sarcomatous transformation. Two cases with a review of the literature.J Neurooncol. 1989; 7: 25-29
- Fibrous dysplasia: rapid malignant transformation into osteogenic sarcoma - A rare occurance.Journal of Pathology of Nepal. 2012; 2: 335-337
- Malignancies in fibrous dysplasia.Cancer. 1994; 73: 1411-1424
- JBI Manual for Evidence Synthesis.JBI. 2020;
- Methodological guidance for systematic reviews of observational epidemiological studies reporting prevalence and cumulative incidence data.Int J Evid Based Healthc. 2015; 13: 147-153
- Methodological quality of case series studies: an introduction to the JBI critical appraisal tool.JBI Evid Synth. 2020; 18: 2127-2133
- Malignant transformation of polyostotic fibrous dysplasia with aberrant keratin expression.Hum Pathol. 2017; 62: 170-174
- Radiological findings in an unusual osteosarcoma in the maxilla.Dentomaxillofac Radiol. 2000; 29: 180-184
- Osteosarcoma mimicking fibrous dysplasia of the jaw.Int J Oral Maxillofac Surg. 1987; 16: 112-115
Article info
Publication history
Published online: December 23, 2022
Accepted:
December 16,
2022
Received in revised form:
November 10,
2022
Received:
April 28,
2022
Identification
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