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Table of Contents
CASE REPORT
Year : 2016  |  Volume : 6  |  Issue : 1  |  Page : 25-28

Prosthodontic rehabilitation of a pediatric patient affected with anhidrotic ectodermal dysplasia: A rare case report


1 Department of Pedodontics and Preventive Dentistry, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, India
2 Department of Periodontology and Oral Implantology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, India

Date of Web Publication10-Aug-2016

Correspondence Address:
Gurlal Singh Brar
Department of Pedodontics and Preventive Dentistry, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2229-5194.188162

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   Abstract 

Ectodermal dysplasia (ED) is a hereditary disease characterized by congenital dysplasia of one or more ectodermal structures and other accessory appendages. The oral manifestations are anodontia and poor bony foundation which impairs both esthetic as well as masticatory function. The prosthodontic management of patients with such dysplastic condition necessitates a multidisciplinary approach. The strong, flexible nature of flexible denture material is perfectly suited to the variety of natural conditions in the mouth, simplifying design and enabling the flexible nylon resin to act as a built-in stress-breaker that provides superior function and stress distribution. Flexible partial dentures certainly offer advantages over conventional partials by way of superior esthetics, better function, durable material, and longevity of the prosthesis. This case report describes the prosthodontic oral rehabilitation of a 14-year-old female pediatric patient with anhidrotic ED using resin flexible prosthesis (Valplast).
Clinical Relevance to Interdisciplinary Dentistry

  • A multidisciplinary approach comprising restorative, prosthetic, and periodontal treatment was done to achieve a satisfactory result. The treatment led to significant improvements in appearance, speech, and masticatory function
  • The focus is on the interdisciplinary management of a patient with complex restorative needs
  • An integrated approach that addresses both skeletal and dental issues can provide superior results. In this case, restorative goals guided the overall treatment plan, requiring the integration of restorative services at several stages of care, which involved a team of pedodontist, prosthodontist, and periodontist
  • Although implants are the ideal choice for partial anodontia, this case was treated in a conservative and minimally invasive method. Eventually the patient was satisfied with the prosthesis as function was re-established and esthetics was not compromised. Long-term success depends on regular recall appointments and meticulous maintenance of oral and prosthetic hygiene.

Keywords: Anodontia, ectodermal dysplasia, prosthodontic rehabilitation


How to cite this article:
Khinda VI, Khinda P, Brar GS, Yadav A. Prosthodontic rehabilitation of a pediatric patient affected with anhidrotic ectodermal dysplasia: A rare case report. J Interdiscip Dentistry 2016;6:25-8

How to cite this URL:
Khinda VI, Khinda P, Brar GS, Yadav A. Prosthodontic rehabilitation of a pediatric patient affected with anhidrotic ectodermal dysplasia: A rare case report. J Interdiscip Dentistry [serial online] 2016 [cited 2019 Dec 14];6:25-8. Available from: http://www.jidonline.com/text.asp?2016/6/1/25/188162


   Introduction Top


Ectodermal dysplasia (ED) is a relatively rare disorder, with a prevalence of 1:100,000 live births and is more frequent in males. [1] It was first described by Thurnam in 1848 and was coined by Weech in 1929. [2]

ED is of two types: Hidrotic ED in which the sweat glands are normal and anhidrotic ED (AED). [3] Since patients with ED have psychosocial issues due to the orofacial manifestations presenting at such young age, restoring appearance and function is more challenging than usual. There are multiple treatment options for this condition, but the most frequent prosthetic treatment of ED in young patients is removable prosthesis.

The uniqueness of this clinical case is the treatment of AED, which is certainly more severe form and is characterized by lack of sweat glands with flexible partial dentures. The strong and flexible nature of the material is perfectly suited to the variety of natural conditions in the mouth, simplifying design and enabling the flexible resin to act as a built-in stress-breaker to provide superior function and stress distribution in a removable partial denture (RPD).

Flexible partial dentures certainly offer advantages over conventional partials by way of superior esthetics, better function, durable material, and longevity of the prosthesis. This clinical report explores the use of flexible denture base material for the fabrication of RPD in a pediatric patient with AED.


   Case report Top


A 14-year-old girl reported with the chief complaint of inability to masticate and unesthetic appearance. She desired replacement of her missing teeth. The patient gave a history of lack of sweating, skin dryness, and raised body temperature during hot weather. Extraoral examination revealed frontal bossing, depressed nasal bridge, prominent supraorbital ridges, protuberant lips. Nails appeared normal. Intraoral examination revealed absence of saliva and dry oral mucosa. Cone-shaped teeth were present in relation to 11, 21. Other teeth present were 55, 65, 16, 26, 27, 36, 46; among these, 55 and 65 were severely decayed and 36, 46 had occlusal caries [Figure 1]a-d. Clinical findings suggested a diagnosis of AED. The treatment was planned into the following four stages:
Figure 1: (a and b) Preoperative frontal view (c) maxillary arch (d) mandibular arch

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  • In the first stage, oral prophylaxis was done
  • The second stage was surgical phase where 55, 65 were extracted under local anesthesia
  • The third stage was restorative and prosthodontic phase. In restorative part, composite build ups were carried out in maxillary anterior to restore their anatomy. In prosthodontic management, both upper and lower flexible dentures were planned. To begin with, an alginate impression was made followed by border molding to record the borders. Then, the dual impression was taken and occlusion rims were fabricated in both maxillary and mandible arches. All the necessary steps including jaw relation, teeth setting, and try-in were performed and after careful evaluation, the maxillary and mandibular partial prostheses were fabricated in Valplast resin in the conventional heat cure acrylic resin. The dentures were then delivered to the patient [Figure 2]a-h
  • The fourth stage was maintenance phase which included periodic follow-ups to monitor oral hygiene, gingival condition, occlusion, and tooth mobility. Future visits were scheduled after every 6 months to monitor bone growth and for denture relining.
Figure 2: (a-h) Steps of fabrication of flexible removable prosthesis

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   Discussion Top


The oral rehabilitation of patients presenting with congenitally missing dentition is challenging because of the need for a multidisciplinary approach. [4] Preservation of alveolar bone is imperative in ED patients because they must depend on the alveolar ridges for prosthesis support from a very early age. Prosthetic treatment modes using RPD, complete denture, and dental implants are the primary treatment options in patient with ED. [5]

The implant-made prosthesis is usually restricted to patients with completed craniofacial growth and is probably best to hold off until adolescence. Implants placed in ED patients younger than 18 years have a higher risk of failure. Implant insertion in children or adolescents can have several unfavorable potential effects including trauma to tooth germs and multidimensional restrictions of skeletal craniofacial growth. For young patients, the use of RPD is a reversible treatment that can significantly improve functions and esthetics without jeopardizing compromised dentitions. [6]

Oral rehabilitation of ED children is necessary to improve both sagittal and vertical skeletal relationship during craniofacial growth and development so as to provide improvements in esthetics, emotional well-being, stomatognathic efficiency, and temporomandibular joint function. [7] Optimal dental treatment should be commenced as soon as possible to avoid possible resorption, atrophy of the alveolar ridges and to control the vertical dimension, which can be severely affected by the total or partial lack of teeth. The prosthetic treatment should be carried out on an individual basis, aimed always toward providing good occlusal stability. It also aids in phonation and mastication. These factors instill greater self-confidence and patient acceptance. [8] Similarly, as in this case, removable partial prosthesis with flexible resin restores the function (mastication), speech, and most importantly facial esthetic which boost up the self-confidence of the patient.

The use of RPD was the ideal treatment option that could significantly improve function and esthetics. Due to the patient's young age, poor oral hygiene, xerostomia, and insufficient quantity of alveolar bone, endosseous implants placement was not possible. The bone height and width was not sufficient for implant insertion. Fixed partial denture was not opted due to oligodontia as rigid fixed partial dentures could interfere with jaw growth, especially if the prosthesis crosses the midline. [9]

Initially, the hard acrylic polymers used in artificial removable dentures were used in combination with chrome cobalt alloys. While conventional metal and acrylic tooth-bearing removable partials are functional, there are a few disadvantages. They are esthetically displeasing to most patients and lead to some psychological hesitation about smiling and speaking. The overall rigidity of the partials causes a wide range of impact damage to the existing bone and tissue structures. Because of this, partials must be relined regularly. Many people have an allergic reaction or slight irritation from one or more of the components of the polymer used in the acrylic base material. [10]

Partials made from the Valplast resin (Valplast International Corp., Westbury, NY, USA) have the advantages that metal and acrylic combination partials do not. These partials are esthetically superior because they are practically invisible. The partials (Valplast) designed correctly do not engage the abutment teeth alone for support and retention. The gentle motion of the partial over the gum tissue produces a massaging effect that can prolong the healthier condition of the gums. Because of this, it is less likely that the patients will come back often to have the partials relined to accommodate deteriorating formations. [6] The technique offers an effective method for constructing partial dentures with certain advantages which are explained as follows: [10]

  • More acceptable esthetics since there are no metal clasps
  • The material has good flexibility like titanium. Therefore, even if there is a little bit of bending, it comes back to the original shape and position
  • Even if there is slight shifting of the remaining teeth over time, the flexibility of the denture material allows the use of prosthesis with little adjustment
  • There is no need of modification of the remaining teeth to receive occlusal rests as for the metal clasps
  • Rebasing (changing the entire plastic/tissue area except the acrylic teeth) is possible.
However, flexible partial denture has certain drawbacks as follows:

  • Being a plastic material, it cannot be made into thin sections like metal. It is likely to break if cut into thin sections
  • Since they need to be made bulkier than cast partials, it may take longer to get used to a flexible partial denture
  • It does not conduct heat and cold like metal. Therefore, the patient may not enjoy certain foods such as hot soup or ice cream
  • Since flexible dentures utilize the gaps (because of some missing teeth) for the "Retento-Grip Tissue-bearing Technique" [11] for retention, the remaining teeth have to be in fairly good periodontal health
  • The patients that have periodontal problem may have several teeth that are mobile due to bone loss. Therefore, the dentulous area keeps on flexing causing unfavorable forces on the present teeth that in turn aggravates the periodontal problem
  • The laboratory fee is a little higher
  • Requires more chair-side time for adjustment
  • Requires special instruments (knives and polishing kit) to make the adjustment
  • A flexible denture is very hard to repair if fractured. No additions can be made onto it. In such cases, rebasing (changing the entire plastic/tissue area except the acrylic teeth) is recommended. [10] Hence, the patient cooperation is a major concern here, especially in case of pediatric patient.

   Conclusion Top


The present clinical report demonstrated that flexible RPDs associated with direct composite restorations could be a reversible, relatively inexpensive method of treatment for ED patients. For the patient described, the treatment improved esthetics, oral functions and established a more favorable plane of occlusion. The patient's social confidence also improved significantly because of the full mouth rehabilitation.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Melilli D, Bortolotti L, Curro G. Prosthetic rehabilitation of a young patient affected by ectodermal dysplasia with the new eclipse resin system. Int J Clin Dent 2012;5:308-17.  Back to cited text no. 1
    
2.
Varghese G, Sathyan P. Hypohidrotic ectodermal dysplasia - A case study. Oral Maxillofac Pathol J 2011;2:123-6.  Back to cited text no. 2
    
3.
Neville BV, Damm DD, Allen CM, Bouquot EJ. Dermatologic diseases. In: Neville BV, editor. Oral and Maxillofacial Pathology. 3 rd ed. Saunders: Philadelphia. Elsevier Publisher; 2009.  Back to cited text no. 3
    
4.
Dinesh NN. Flexible denture for hereditary ectodermal dysplasia. Clin Dent 2012;6:52-6.  Back to cited text no. 4
    
5.
Aydinbelge M, Gumus HO, Sekerci AE, Demetoglu U, Etoz OA. Implants in children with hypohidrotic ectodermal dysplasia: An alternative approach to esthetic management: Case report and review of the literature. Pediatr Dent 2013;35:441-6.  Back to cited text no. 5
    
6.
Jain N, Naitam D, Wadkar A, Nemane A, Katoch S, Dewangan A. Prosthodontic rehabilitation of hereditary ectodermal dysplasia in an 11-year-old patient with flexible denture: A case report. Case Rep Dent 2012;2012:489769.  Back to cited text no. 6
    
7.
Prithviraj DR, Harshamayi P, Madan V, Kumar GC, Shruthi DP. A complete prosthodontic solution for patient with ectodermal dysplasia: A review. International Journal of Therapeutic Applications 2014;17:16-20.  Back to cited text no. 7
    
8.
Shigli A, Reddy RP, Hugar SM, Deshpande D. Hypohidrotic ectodermal dysplasia: A unique approach to esthetic and prosthetic management: A case report. J Indian Soc Pedod Prev Dent 2005;23:31-4.  Back to cited text no. 8
[PUBMED]  Medknow Journal  
9.
Pigno MA, Blackman RB, Cronin RJ Jr., Cavazos E. Prosthodontic management of ectodermal dysplasia: A review of the literature. J Prosthet Dent 1996;76:541-5.  Back to cited text no. 9
    
10.
Thakral GK, Aeran H, Yadav B, Thakral R. Flexible partial dentures - A hope for the challenged mouth. Peoples J Sci Res 2012;5:55-9.  Back to cited text no. 10
    
11.
Iselin W, Meier C, Lufi A, Lutz F. The flexible gingival epithesis. The practical procedure, laboratory technics and clinical experience. Schweiz Monatsschr Zahnmed 1990;100:966-79.  Back to cited text no. 11
    


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