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Follow-up CT findings of recurrent familial gigantiform cementoma of a female child

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Abstract

Familial gigantiform cementoma (FGC) is a rare autosomal dominant, benign fibro-cemento-osseous lesion of the jaws that can cause severe facial deformity. True FGC with familial history is extremely rare and there has been no literature regarding the radiological follow-up of FGC. We report a case of recurrent FGC in an Asian female child who has been under our observation for 6 years since she was 15 months old. After repeated recurrences and subsequent surgeries, the growth of the tumor had seemed to plateau on recent follow-up CT images. The transition from an enhancing soft tissue lesion to a homogeneous bony lesion on CT may indicate decreased growth potential of FGC.

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References

  1. Cannon JS, Keller EE, Dahlin DC. Gigantiform cementoma: report of two cases (mother and son). J Oral Surg. 1980;38:65–70.

    PubMed  CAS  Google Scholar 

  2. Young SK, Markowitz NR, Sullivan S, Seale TW, Hirschi R. Familial gigantiform cementoma: classification and presentation of a large pedigree. Oral Surg Oral Med Oral Pathol. 1989;68:740–7.

    Article  PubMed  CAS  Google Scholar 

  3. Oikarinen K, Altonen M, Happonen RP. Gigantiform cementoma affecting a Caucasian family. Br J Oral Maxillofax Surg. 1991;29:194–7.

    Article  CAS  Google Scholar 

  4. Coleman H, Altini M, Kieser J, Nissenbaum M. Familial florid cemento-osseous dysplasia: a case report and review of literature. J Dent Assoc S Afr. 1996;51:766–70.

    PubMed  CAS  Google Scholar 

  5. Finical SJ, Kane WJ, Clay RP, Bite U. Familial gigantiform cementoma. Plast Reconstr Surg. 1999;103:949–54.

    Article  PubMed  CAS  Google Scholar 

  6. Rossbach HC, Letson D, Lacson A, Ruas E, Salazar P. Familial gigantiform cementoma with brittle bone disease, pathologic fractures and osteosarcoma: a possible explanation of an ancient mystery. Pediatr Blood Cancer. 2005;44:390–6.

    Article  PubMed  Google Scholar 

  7. Han WJ, Kim EK. Familial gigantiform cementoma. Korean J Oral Maxillofac Radiol. 2006;36:157–62.

    Google Scholar 

  8. Neville BW, Damm D, Allen CM, Bouquot JE. Oral and maxillofacial pathology. 3rd ed. Philadelphia: Saunders; 2009. pp. 645–46.

    Google Scholar 

  9. Abdelsayed RA, Eversole LR, Singh BS, Scarbrough FE. Gigantiform cementoma: clinicopathologic presentation of 3 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endo. 2001;91:438–44.

    Article  CAS  Google Scholar 

  10. Mahomed F, Altini M, Meer S, Coleman H. Cemento-osseous dysplasia with associated simple bone cyst. J Oral Maxillofac Surg. 2003;63:1549–54.

    Article  Google Scholar 

  11. Williams HK, Mangham C, Speight PM. Juvenile ossifying fibroma: an analysis of eight of eight cases and a comparison with other fibro-osseous lesions. J Oral Pathol Med. 2000;29:13–8.

    Article  PubMed  CAS  Google Scholar 

  12. Von Wowern N. Cherubism: A 36-year long time follow-up of 2 generations in different families and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2000;90:765–72.

    Article  Google Scholar 

  13. Kapadia SB, Frisman DM, Hitchcock CL, Ellis GL, Popek EJ. Melanotic neuroectodermal tumor of infancy. Clinicopathological, immunohistochemical and flowcytometric study. Am J Surg Pathol. 1993;17:566–73.

    Article  PubMed  CAS  Google Scholar 

  14. MacDonald-Jankowski DS. Florid cemento-osseous dysplasia: a systematic review. Dentomaxillofac Radiol. 2003;32:141–9.

    Article  PubMed  CAS  Google Scholar 

  15. Melrose RJ, Abrams AM, Mills BG. Florid osseous dysplasia: a clinical-pathologic study of thirty-four cases. Oral Surg Oral Med Oral Pathol. 1976;41:62–82.

    Article  PubMed  CAS  Google Scholar 

  16. Takeda Y, Fujioka Y. Multiple cemento-ossifying fibroma. Int J Oral Maxillofac Surg. 1987;16:368–71.

    Article  PubMed  CAS  Google Scholar 

  17. Bradley Jr ES, Leake D. Ossifying fibroma involving the maxilla and the mandible; report of a case. Oral Surg Oral Med Oral Pathol. 1968;26:605–14.

    Article  PubMed  Google Scholar 

  18. Hauser MS, Freije S, Payne RW, Timen S. Bilateral ossifying fibroma of the maxillary sinus. Oral Surg Oral Med Oral Pathol. 1989;68:759–63.

    Article  PubMed  CAS  Google Scholar 

  19. Thompson SH, Altini M. Gigantiform cementoma of the jaws. Head Neck. 1989;11:538–44.

    Article  PubMed  CAS  Google Scholar 

  20. Chacon GE, Ugalde CM, Jabero MF. Genetic disorders and bone affecting the craniofacial skeleton. Oral Maxillofac Surg Clin North Am. 2007;19:467–74.

    Article  PubMed  Google Scholar 

  21. Moshref M, Khojasteh A, Kazemi B, Roudsari MV, Varshowsaz M, Eslami B. Autosomal dominant gigantiform cementoma associated with bone fractures. Am J Med Genet A. 2008;146A:644–8.

    Article  PubMed  Google Scholar 

  22. Levin LS, Wright JM, Byrd DL, Greenway G, Dorst JP, Irani RN, et al. Osteogenesis imperfecta with unusual skeletal lesions: report of three families. Am J Med Genet. 1985;21:257–69.

    Article  PubMed  CAS  Google Scholar 

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Acknowledgement

This study was supported by the Overseas Research Program of Seoul National University Dental Hospital.

Conflict of Interest

The authors declare that they have no conflict of interest.

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Authors and Affiliations

  1. Department of Oral and Maxillofacial Radiology, School of Dentistry, Seoul National University, 101 Daehak-ro, Jongnogu, Seoul, 110-768, Republic of Korea

    Saiquat Shah, Kyung-Hoe Huh, Won-Jin Yi, Min-Suk Heo, Sam-Sun Lee & Soon-Chul Choi

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  1. Saiquat Shah
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  2. Kyung-Hoe Huh
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  3. Won-Jin Yi
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  4. Min-Suk Heo
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Correspondence to Kyung-Hoe Huh.

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Shah, S., Huh, KH., Yi, WJ. et al. Follow-up CT findings of recurrent familial gigantiform cementoma of a female child. Skeletal Radiol 41, 341–346 (2012). https://doi.org/10.1007/s00256-011-1245-9

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  • DOI: https://doi.org/10.1007/s00256-011-1245-9

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