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Original Research

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A Rare Case of Gingival Fibromatosis Associated with Hypertrichosis and a Dysmorphic Face

  • Parimala Kulkarni1,*,
  • Neeraj Agrawal1
  • Sanjeev Tyagi1
  • Halaswamy Kambalimath1

1Department of Pediatric and Preventive Dentistry, People’s Dental Academy, Bhopal.(MP), India.

DOI: 10.17796/jcpd.35.3.a1731m1l2812lvm6 Vol.35,Issue 3,May 2011 pp.305-308

Published: 01 May 2011

*Corresponding Author(s): Parimala Kulkarni E-mail: drtyagip@gmail.com

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Abstract

Several forms of hypertrichosis have been described with and without gingival hyperplasia; some of them are recognized as genetic disorder and associated with syndromes. In all reported cases the most striking differences from other are the craniofacial features. We present a case of a 6-year-old boy with hypertrichosis associated with gingival hyperplasia and a characteristic, coarse face and we consider this case to be a distinctive entity.

Keywords

congenital hypertrichosis, gingival fibromatosis, children.

Cite and Share

Parimala Kulkarni,Neeraj Agrawal,Sanjeev Tyagi,Halaswamy Kambalimath. A Rare Case of Gingival Fibromatosis Associated with Hypertrichosis and a Dysmorphic Face. Journal of Clinical Pediatric Dentistry. 2011. 35(3);305-308.

URL:

https://www.jocpd.com/articles/10.17796/jcpd.35.3.a1731m1l2812lvm6

References

1. Gross S D.Case of hypertrophy of the gums. Louis-Ville Rev, 1: 232–237, 1856.

2. Bondenson J, Miles AEW. Julia Pastrand, the non descript: an example of congenital, generalized hypertrichosis terminalis with gingival hyperplasia. Am J Med Genet, 47: 198–212, 1993.

3. Winter G, Simpkiss M. hypertrichosis with hereditary gingival hyperplasia. Arch Dis Child, 49: 394–399, 1974.

4. Baumeister FAM, Schwarz HP, Stengel-Rutkowsky S. child hood hypertrichosis, diagnosis and management. Arch Dis Child, 72: 457–459, 1995.

5. Witkop, C.J.heterogeneity in gingival fibromatosis birth defects. original actual series. vol 7, part II, Williams and Wilkins, Baltimore; 210, 1971.

6. Baumeister FAM, Egger J, Schildhauer MT, Stengel-Rutkowski S. Ambras syndrome: delineation of a unique hypertrichosis universalis congenita and association with a balanced pericentric inversion(8)(p11.2;q22). Clin Genet, 44: 121–128, 1993.

7. Cantu J, Figuera L.Ambras syndrome and congenital generalized hypertrichosis. Clin Genet, 46: 384, 1994.

8. Baumeister FAM, Stengel-Rutkowsky S. differentiation of congenital hypertrichosis from Ambras Syndrome Clin Genet, 46: 441, 1994.

9. Cantu JM, Garcia-Cruz D, Sanchez-Corona J, Hernandez A, Nazara Z. A distinct osteochondrodysplasia with hypertrichosis individualization of a probable autosomal recessive entity Hum Genet, 60: 36–41, 1982.

10. Garcia-Cruz D, Sanchez-Corona J, Nazara Z et al. Congenital hypertrichosis, osteochondrodysplasia, and cardiomegaly: further delineation of a new genetic syndrome. Am J Med Genet 69: 138–151, 1997.

11. Canun S et al. hypertrichosis terminalis, gingival hyperplasia and a characteristic face:a new dictinct entity. Am J Med Genet, 116A: 278–283, 2003.

12. Anderson J, Cunliffe WJ, Roberts DF, and Close H. Hereditary gingival fibromatosis. British Medical Journal, 3: 218, 1969.

13. Rook A, Wilkinson DS, Ebling FJG, Champion RM, Burttan JL. The hair In: Ebling FJG, Drawleer R, Rook A,eds. Textbook of Dermatology, 4th ed. Oxford:Blackwell, 1986:1959.

14. Bertollins AT, Freedbery IM. Hair In: Fitz Patrick TB, Eisen AZ, Wolff K, Freedberg IM , Austen KF, eds. Dermatology in general medicine, 3rd ed. Newyork: Mc Grow-Hill: 645–646, 1987.

15. Mocias-Floias MA, Garcia Cruz D, Rivera H, Escobar-Lujan M, Melendez Vega A, Rivas Campos D, Rodiquez-Collazo F, MorenoArellano I, Cantu JM. A new form of hypertrichosis inherited as an x linked dominant trait. Hum Genet, 66: 66–70, 1984.

16. Horning GM, Fisher JG, Baker BF, et al gingival fibromatosis with hypertrchosis. J Periodontal 56: 344–347, 1985. 17. Carnero RC Bornancini CA. Hereditary generalized gingival fibromatosis associated with hypertrichosis. J Oral Maxillofacial Surg, 46: 415–520, 1988.

18. Anavi Y, jerman P, Mintz S, Kiviti S. Idiopathic familial gingival fibromatosis associated with mental retardation, epilepcy and hypertrichosis. Dev. Med Child Neural, 31: 538–542, 1989.

19. Kiss P. gingival fibromatosis, mental retardation, epilepcy, and hypertrichosis. Dev(Med) Child Neural, 32: 459–460, 1990.

20. Jorgenson RJ, Cocker ME, variation in inheritance and expression of gingival fibromatosis. J Periodontol, 45: 472, 1974.

21. Sun M, Li N, Dong W, Chen Z, Liu Q, Xu Y et al. Copy-number mutations on chromosome17q24.2-q24.3 in congenital generalized hypertrichosis terminalis with or without gingival hyperplasia. Am J Hum Genet, 84: 807–813, 2009.

22. Ravin JG, Hodge GP 1969. Hypertrichosis portrayed in art. JAMA, 207: 533–53522, 1969.

23. Freire-Maria, Felizali J., de Figuereid AC, Opitz JM, Parreira M, Maia NA. Hypertrichosis lanuginose in a mother and son. Clin Genet, 10: 303–306, 1976.

24. Balducci R,Toscano V, Tedeschi B, Mangiantini A, Toscano R., Galasso C., Cianfacani S., Boscherini B. A new case of Ambras syndrome associated with a paracentric inversion (8)(q12;q22) Clin Genet, 53: 466–468, 1998.

25. Irvin DA, Dolan MO, Hadden DR, Stewart FJ, Bingham EA, Nevin NC. An autosomal dominant syndrome of acromegaloid facial appearance and generalized hypertrichosis terminalis, J.Med Genet, 33: 972–974, 1996.

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