Download PDF
CC BY-NC-ND 4.0 · Eur J Dent 2011; 05(04): 486-491
DOI: 10.1055/s-0039-1698923
Original Article
Dental Investigation Society

Treatment of Cherubism with Salmon Calcitonin: A Case Report

Osman A Etoz
a   Erciyes University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Kayseri, Turkey.
,
Dogan Dolanmaz
b   Selcuk University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Konya, Turkey.
,
Omer Gunhan
c   Gülhane Military Medical Academy and Medical Faculty, Department of Pathology, Ankara, Turkey.
› Author Affiliations
Further Information

Publication History

Publication Date:
18 March 2020 (online)

Also available at
  PDF Download Permissions and Reprints

ABSTRACT

Cherubism is a familial disease of the jaws which is inherited via autosomal dominant manner. Typical features of cherubism include a painless bilateral, symmetrical enlargement of the jaws, misalignment of teeth, and intra-osseous central giant cell granuloma-like lesions, which are usually evident in early childhood. Treatment of cherubism consists of local curettage of the lesions, jaw contouring, intralesional steroid injections, and systemic calcitonin administration as well. Calcitonin therapy for central giant cell granuloma of the jaws is well documented, and favorable results have been achieved. However, fewer reports have been presented in regard to calcitonin administration for cherubism. In the present report, a 14-year-old boy with cherubism who had intra-osseous cherubic lesions in his mandible was treated with an administration of 200 IU systemic calcitonin every other day via his nasal passage for duration of more than two years. After 30 months of calcitonin therapy, the lesions in the mandible were significantly regressed, and calcitonin application was ceased. Despite some drawbacks, such as unpredictable efficient absorption and patient tolerability, nasal administration is the easiest way to use calcitonin therapy on children. In this report, every-other-day applications of calcitonin increased patient tolerability and might be considered as an effective treatment for mild cherubic lesions. (Eur J Dent 2011;5:486-491)

Key words:

Cherubism - Salmon calcitonin - Central giant cell granuloma
 

  • REFERENCES

  • 1 Jones WA. Familial multilocular cystic disease of the jaws. Am J Cancer 1933;17:946.
  • 2 De Lange J, van den Akker H, Scholtemeijer M. Cherubism treated with calcitonin: report of a case. J Oral Maxillofac Surg 2007;65:1665-1667.
  • 3 Anderson DE, McClendon JL. Cherubism-hereditary fibrous dysplasia of the jaws; I. genetic considerations. Oral Surg Oral Med Oral Pathol 1962;15(suppl.2):5-16.
  • 4 De Lange J, van Maarle M, van den Akker HP, Redeker EJW. A new mutation in the SH3BP2 gene showing reduced penetrance in a family affected with cherubism. Oral Surg Oral Med Oral Pathol 2007;103:378-381.
  • 5 Gunhan O. Oral ve Maksillofasiyal Patoloji (1st edn). Ankara: Atlas Kitapcilik; 2001.
  • 6 Carlos R, Sedano HO. Intralesional corticosteriods as an alternative treatment for central giant cell granuloma. Oral Surg Oral Med Oral Pathol 2002;93:161-166.
  • 7 Kaban LB, Mulliken JB, Ezekowitz RA et al. Antiangiogenic therapy of a recurrent giant cell tumor of the mandible with interferon alfa-2a. Pediatrics 1999;103:1145-1149.
  • 8 Harris M. Central giant cell granulomas of the jaws regress with calcitonin therapy. Br J Oral Maxillofac Surg 1993;31:89-94.
  • 9 Pogrel MA. Calcitonin therapy for central giant cell granuloma. J Oral Maxillofac Surg 2003;61:649-653.
  • 10 Von Wowern N. Cherubism: a 36-year long term follow-up of 2 generations in different families and review of the literature. Oral Surg Oral Med Oral Pathol 2000;90:765-772.
  • 11 Hyckel P, Berndt A, Schleier P, Clement JH, Beensen V, Peters H, et al. Cherubism – new hypotheses on pathogenesis and therapeutic consequences. J Craniomaxillofac Surg 2005;33:61-68.
  • 12 Grünebaum M, Tiqva P. Non-familial cherubism: report of a case. J Oral Surg 1973;31:632-635.
  • 13 Kozakiewicz M, Perezynska-Partyka W, Kobos J. Cherubism – clinical picture and treatment. Oral Diseases 2001;7:123-130.
  • 14 Pogrel MA, Regezi JA, Harris ST, Goldring SR. Calcitonin treatment for central giant cell granulomas of the mandible: report of two cases. J Oral Maxillofac Surg 1999;57:848- 853.
  • 15 Flanagan AM, Nui B, Tinkler SMB, Horton MA, Williams DM, Chambers TJ. The multinucleate cell in giant cell granulomas of the jaw are osteoclast. Cancer 1988;62:1139-1145.
  • 16 Southgate J, Sarma U, Townend JV, Barron J, Flanagan AM. Study of the cell biology and chemistry of cherubism. J Clin Pathol 1998;51:831-837.
  • 17 Lannon DA, Earley MJ. Cherubism and its charlatans. Br J Plast Surg 2001;54:708-731.
  • 18 Hart W, Schweitzer DH, Slootweg PJ, et al. Man with cherubism. Ned Tijdschr Geneeskd 2000;144:34-38.
  • 19 Motamedi MHK. Treatment of cherubism with locally aggressive behavior presenting in adulthood: report of four cases and a proposed new grading system. J Oral Maxillofac Surg 1998;56:1336-1342.
  • 20 Seward GR, Hankey GT. Cherubism. Oral Surg Oral Med Oral Pathol 1957;10:952-974.
  • 21 Azria M. The calcitonines, physiology and pharmacology. London: Karger;1989.
  • 22 De Lange J, Rosenberg AJWP, van den Akker HP, Koole R, Wirds JJ, van den Berg. Treatment of giant cell granuloma with calcitonin. Int J Oral Maxillofac Surg 1999;28:372-376.