SciELO - Scientific Electronic Library Online

 
vol.9 número1 índice de autoresíndice de assuntospesquisa de artigos
Home Pagelista alfabética de periódicos  

Brazilian Journal of Oral Sciences

versão On-line ISSN 1677-3225

Braz. J. Oral Sci. vol.9 no.1 Piracicaba Jan./Mar. 2010

 

CASE REPORT

 

Prosthetic rehabilitation for a patient with hypohidrotic ectodermal dysplasia: a clinical case

 

 

Guna ShekharI; Alluri RamaRajuII; Chandrasekhar RaoIII; SaradaI

IAssociate Professor, Faculty of Pediatric Dentistry, VDC, NTR University of Health Sciences, AP, India
IIProfessor, Faculty of Prosthetic Dentistry, VDC, NTR University of Health Sciences, AP, India
IIIProfessor, Faculty of Pediatric Dentistry, VDC, NTR University of Health Sciences, AP, India

Correspondence

 

 


ABSTRACT

Hypohidrotic ectodermal dysplasia (HED) is a hereditary syndrome, characterized by a classic triad of hypotrichosis, hypodontia and hypohidrosis. The case of an 8-year-old girl with HED presenting oligodontia and marked resorption of maxillary and mandibular alveolar ridges is reported. A specially designed aluminum wire tray was used for taking impressions of both dental arches. Prosthetic rehabilitation included a removable maxillary overdenture and a mandibular conventional acrylic resin denture made tissue-compatible by means of a soft denture liner attached to denture base. The proposed treatment plan provided improved esthetics, function, and quality of life.

Keywords: ectodermal dysplasia, oligodontia, soft liner, mucosa supported dentures.


 

 

Full text available only in PDF format.

 

 

References

1. Neville BW Oral and maxillofacial pathology. Philadelphia: Saunders; 1995. p.542-3.         [ Links ]

2. Della Valle D, Chevitarese AB, Maia LC, Farinhas JA. Alternative rehabilitation treatment for a patient with ectodermal dysplasia. J Clin Pediatr Dent. 2004; 28: 103-6.         [ Links ]

3. Pinheiro M, Freire-Maria N. Ectodermal dysplasias: a clinical classification and a casual review. Am J Med Genet. 1994; 53: 153-62.         [ Links ]

4. Kargul B, Alen TK, Alabay U, Atasu M. Hypohidrotic ectodermal dysplasia: Dental clinical genetic and dermatoglyphic findings of three cases. J Clin Pediatr Dent. 2001; 26: 5-12.         [ Links ]

5. Ohno K, Ohmori I. Anodontia with hypohidrotic ectodermal dysplasia in young female: a case report. Pediatr Dent. 1999; 22: 49-52.         [ Links ]

6. Abadi B, Herren C. Clinical treatment of ectodermal dysplasia: A case report. Quintessence Int. 2001; 32: 743-45.         [ Links ]

7. Gorlin RJ, Cohen MM, Hennekam RC. Hypohidrotic ectodermal dysplasia. In: Syndromes of the Head and Neck. 4th ed. Oxford: Oxford University Press; 2001. p.540 5.         [ Links ]

8. Kere J, Srivastava AK, Montonen O, Zonana J, Thomas N, Ferguson B, ET al. X-linked anhidrotic (hypohidrotic) ectodermal dysplasia is caused by mutation in a novel transmembrane protein. Nat Genet. 1996; 13: 409-16.         [ Links ]

9. Huang C, Yang Q, Ke T, Wang H, Wang X, Shen J et al. A novel de novo frame-shift mutation of the EDA gene in a Chinese Han family with hypohidrotic ectodermal dysplasia. J Hum Genet. 2006; 51: 1133-7.         [ Links ]

10. Fan Huali, Ye Xiaogian, Shi Lisong, Yin W, Hua B, Song G et al. Mutations in the EDA gene are responsible for X-linked hypohidrotic ectodermal dysplasia and hypodontia in Chinese kindreds. Eur J Oral Sci. 2008; 116: 412-7.         [ Links ]

11. Van der Hout AH, Oudesluijs GG, Venema A, Verheij JB, Mol BG, Rump P et al. Mutation screening of the ectodysplasin-A receptor gene EDAR inhypohidrotic ectodermal dysplasia. Eur J Hum Genet. 2008; 16: 673-9.         [ Links ]

12. Guckes AD, Mc Carthy GR, Brahim J. Use of endosseous implants in a 3-year-old child with ectodermal dysplasia: case report and 5-year follow-up. Pediatr Dent. 1997; 19: 282-5.         [ Links ]

13. Hummel P, Guddack S. Psychosocial stress and adaptive functioning in children and adolescents suffering from hypohidrotic ectodermal dysplasia. Pediatr Dermatol. 1997; 14: 180-5.         [ Links ]

14. Granger ER. Practical procedures in oral rehabilitation. Philadelphia: Lippincott; 1962. p.33.         [ Links ]

15. Duncan JD, Caswell CW, Rommerdale EH. Simplified technique for placement of a medical-grade adhesive silicone denture liner. J Prosthet Dent. 1985; 54: 599-602.         [ Links ]

16. Franchi L, Branch R, Tollaro I. Craniofacial changes following early prosthetic treatment in a case of hypohidrotic ectodermal dysplasia with complete anodontia. J Dent Child. 1995; 65: 116-72.         [ Links ]

 

 

Correspondence to:
Guna Shekhar
c/o M.Prakash, 160, 5th Cross,
28th main, sector-1, HSR Layout,
Bangalore-102, INDIA
E-mail: drguna1234@yahoo.com

Received for publication: December 04, 2009
Accepted: March 04, 2010