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CC BY-NC-ND 4.0 · Arch Plast Surg 2023; 50(01): 049-053
DOI: 10.1055/s-0042-1758468
Pediatric/Craniomaxillofacial/Head & Neck
Case Report

Psammomatoid Juvenile Ossifying Fibroma of the Maxilla Misdiagnosed as Fibrous Dysplasia: A Clinicopathologic Case Report

Jong-Ho Kim 
1   Department of Plastic and Reconstructive Surgery, Seoul National University College of Medicine, Seoul National University Bundang Hospital, Republic of Korea
,
Jiwon Kang 
1   Department of Plastic and Reconstructive Surgery, Seoul National University College of Medicine, Seoul National University Bundang Hospital, Republic of Korea
,
Seong-ik Kim 
2   Department of Pathology, Seoul National University College of Medicine, Seoul, Republic of Korea
,
Byung Jun Kim 
3   Department of Plastic and Reconstructive Surgery, Seoul National University College of Medicine, Seoul National University Hospital, Republic of Korea
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Abstract

Juvenile ossifying fibroma (JOF) is a variant of the ossifying fibroma and includes two histopathological subtypes: trabecular and psammomatoid. Psammomatoid JOF (PJOF) in craniofacial structures should be distinguished from other fibro-osseous lesions, such as fibrous dysplasia (FD), considering the difference in the treatment protocols. Here, we present a rare case of PJOF that was initially misdiagnosed as a case of FD and emphasize the importance of considering JOF in the differential diagnosis of patients with craniofacial fibro-osseous lesions. A 4-year-old boy demonstrated progressive enlargement of the zygomaticomaxillary area on his left side for the last 6 months. The patient was diagnosed as a case of FD based on the clinical features and radiographic findings, and was operated considering the rapid progression. To achieve facial symmetry, contouring of the zygomatic bone and arch was performed. However, the patient demonstrated rapid enlargement at the 3-month postoperative follow-up. The decision was made to surgically remove the tumor due to visual field impairment. Intraoperatively, a rubbery mass, which was separated from the surrounding cortical bone, was identified and excised. The lesion was confirmed as PJOF by histopathological examination. The possibility of PJOF should not be ruled out in the differential diagnosis of patients with fibrous-osseous lesions. In the event of suspected PJOF, accurate diagnosis should be made through definitive biopsy.

Keywords

psammomatoid juvenile ossifying fibroma - fibrous dysplasia - fibro-osseous lesions - surgical treatment - differential diagnosis

Authors' Contributions

JH Kim and BJ Kim conceptualized the study. JH Kim, J Kang, and S Kim were involved in data curation. JH Kim, J Kang, and S Kim did formal analysis. JH Kim was involved in methodology. BJ Kim did project administration. JH Kim and J Kang contributed to visualization. JH Kim was involved in writing—original draft. JH Kim, J Kang, and BJ Kim reviewed and edited the manuscript.


Ethical Approval

Ethical approval for this study was obtained from the Institutional Review Board in Seoul National University Hospital (IRB no.: H-2206-047-1330)


Patient Consent

Patients provided written informed consent for the publication and the use of their images.




Publication History

Received: 19 February 2022

Accepted: 17 August 2022

Article published online:
06 February 2023

© 2023. The Korean Society of Plastic and Reconstructive Surgeons. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)

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  • References

  • 1 Brannon RB, Fowler CB. Benign fibro-osseous lesions: a review of current concepts. Adv Anat Pathol 2001; 8 (03) 126-143
    CrossrefPubMedGoogle Scholar
  • 2 El-Mofty S. Psammomatoid and trabecular juvenile ossifying fibroma of the craniofacial skeleton: two distinct clinicopathologic entities. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002; 93 (03) 296-304
    CrossrefPubMedGoogle Scholar
  • 3 Margo CE, Ragsdale BD, Perman KI, Zimmerman LE, Sweet DE. Psammomatoid (juvenile) ossifying fibroma of the orbit. Ophthalmology 1985; 92 (01) 150-159
    CrossrefPubMedGoogle Scholar
  • 4 Ranganath K, Kamath SM, Munoyath SK, Nandini HV. Juvenile psammomatoid ossifying fibroma of maxillary sinus: case report with review of literature. J Maxillofac Oral Surg 2014; 13 (02) 109-114
    CrossrefPubMedGoogle Scholar
  • 5 Nair SN, Kini R, Rao PK. et al. Fibrous dysplasia versus juvenile ossifying fibroma: a dilemma. Case Rep Dent 2016; 2016: 6439026
    PubMedGoogle Scholar
  • 6 Toyosawa S, Yuki M, Kishino M. et al. Ossifying fibroma vs fibrous dysplasia of the jaw: molecular and immunological characterization. Mod Pathol 2007; 20 (03) 389-396
    CrossrefPubMedGoogle Scholar
  • 7 Kruse A, Pieles U, Riener MO, Zunker Ch, Bredell MG, Grätz KW. Craniomaxillofacial fibrous dysplasia: a 10-year database 1996-2006. Br J Oral Maxillofac Surg 2009; 47 (04) 302-305
    CrossrefPubMedGoogle Scholar
  • 8 Boyce AM, Burke A, Cutler Peck C, DuFresne CR, Lee JS, Collins MT. Surgical management of polyostotic craniofacial fibrous dysplasia: long-term outcomes and predictors for postoperative regrowth. Plast Reconstr Surg 2016; 137 (06) 1833-1839
    CrossrefPubMedGoogle Scholar
  • 9 Park JW, Jung JH, Park SJ, Lim SY. Evaluation of natural growth rate and recommended age for shaving procedure by volumetric analysis of craniofacial fibrous dysplasia. Head Neck 2020; 42 (10) 2863-2871
    CrossrefPubMedGoogle Scholar
  • 10 Hart ES, Kelly MH, Brillante B. et al. Onset, progression, and plateau of skeletal lesions in fibrous dysplasia and the relationship to functional outcome. J Bone Miner Res 2007; 22 (09) 1468-1474
    CrossrefPubMedGoogle Scholar
  • 11 Malathi N, Radhika T, Thamizhchelvan H. et al. Psammomatoid juvenile ossifying fibroma of the jaws. J Oral Maxillofac Pathol 2011; 15 (03) 326-329
    CrossrefPubMedGoogle Scholar
  • 12 Park S, Lee BJ, Lee JH. et al. Juvenile ossifying fibroma: a clinicopathologic study of 8 cases and comparison with craniofacial fibro-osseous lesions. Korean J Pathol 2007; 41: 373-379
    Google Scholar
  • 13 Linhares P, Pires E, Carvalho B, Vaz R. Juvenile psammomatoid ossifying fibroma of the orbit and paranasal sinuses. A case report. Acta Neurochir (Wien) 2011; 153 (10) 1983-1988
    CrossrefPubMedGoogle Scholar
  • 14 Han J, Hu L, Zhang C. et al. Juvenile ossifying fibroma of the jaw: a retrospective study of 15 cases. Int J Oral Maxillofac Implants 2016; 45 (03) 368-376
    CrossrefPubMedGoogle Scholar
  • 15 Alawi F. Benign fibro-osseous diseases of the maxillofacial bones. A review and differential diagnosis. Am J Clin Pathol 2002; 118 (Suppl): S50-S70
    PubMedGoogle Scholar
  • 16 Eversole R, Su L, ElMofty S. Benign fibro-osseous lesions of the craniofacial complex. A review. Head Neck Pathol 2008; 2 (03) 177-202
    CrossrefPubMedGoogle Scholar
  • 17 Osunde O, Iyogun C, Adebola R. Juvenile aggressive ossifying fibroma of the maxilla: a case report and review of the literature. Ann Med Health Sci Res 2013; 3 (02) 288-290
    CrossrefPubMedGoogle Scholar
  • 18 Gupta D, Garg P, Mittal A. Computed tomography in craniofacial fibrous dysplasia: a case series with review of literature and classification update. Open Dent J 2017; 11: 384-403
    CrossrefPubMedGoogle Scholar
  • 19 Turin SY, Purnell C, Gosain AK. Fibrous dysplasia and juvenile psammomatoid ossifying fibroma: a case of mistaken identity. Cleft Palate Craniofac J 2019; 56 (08) 1083-1088
    CrossrefPubMedGoogle Scholar
  • 20 Voytek TM, Ro JY, Edeiken J, Ayala AG. Fibrous dysplasia and cemento-ossifying fibroma. A histologic spectrum. Am J Surg Pathol 1995; 19 (07) 775-781
    PubMedGoogle Scholar