Journal of Pharmacy And Bioallied Sciences
Journal of Pharmacy And Bioallied Sciences Login  | Users Online: 704  Print this pageEmail this pageSmall font sizeDefault font sizeIncrease font size 
    Home | About us | Editorial board | Search | Ahead of print | Current Issue | Past Issues | Instructions | Online submission




 
 Table of Contents  
DENTAL SCIENCE - CASE REPORT
Year : 2013  |  Volume : 5  |  Issue : 5  |  Page : 128-130  

Multidisciplinary approach of ectodermal dysplasia with implant retained fixed prosthesis


1 Department of Orthodontics, Thai Moogambigai Dental College and Hospital, Chennai, Tamil Nadu, India
2 Department of Oral Surgery, Thai Moogambigai Dental College, Chennai, Tamil Nadu, India
3 Department of Prosthodontics, Karpaga Vinayaga Dental College and Hospital, Chennai, Tamil Nadu, India

Date of Submission02-May-2013
Date of Decision04-May-2013
Date of Acceptance04-May-2013
Date of Web Publication13-Jun-2013

Correspondence Address:
Vishnu Priya
Department of Orthodontics, Thai Moogambigai Dental College and Hospital, Chennai, Tamil Nadu
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-7406.113313

Rights and Permissions
   Abstract 

Ectodermal dysplasia represents a group of rare inherited conditions in which two or more ectodermally derived anatomical structures fail to develop. Early dental intervention can improve patient's appearance, thereby minimizing associated emotional and psychological problems in these patients. Treatment requires a teamwork by medical personnel along with dental professionals of various specialties. Here, a rare case of a young female patient is presented with prosthetic management with implant supported fixed partial denture.

Keywords: Ectodermal, fixed partial denture, implant


How to cite this article:
Priya V, Srivatsa, Ramachandraprabakar, Kannan K, Dwaragesh. Multidisciplinary approach of ectodermal dysplasia with implant retained fixed prosthesis. J Pharm Bioall Sci 2013;5, Suppl S1:128-30

How to cite this URL:
Priya V, Srivatsa, Ramachandraprabakar, Kannan K, Dwaragesh. Multidisciplinary approach of ectodermal dysplasia with implant retained fixed prosthesis. J Pharm Bioall Sci [serial online] 2013 [cited 2019 Oct 23];5, Suppl S1:128-30. Available from: http://www.jpbsonline.org/text.asp?2013/5/5/128/113313

Ectodermal dysplasia (ED) is defined by National Foundation for Ectodermal Dysplasia as a large heterogeneous group of inherited disorder that is defined by primary defect in the development of two or more tissues derived from embryonic ectoderm. [1] Thurnam published the first report of a patient with ED in 1848, [2] but the term was not coined until 1929 [3] by Weech. It is congenital diffuse and nonprogressive disorder. It is of two types: Hypoanhidrotic ED (HAED) X-linked and hypohidrotic ED (HED). [4] ED of the hypohidrotic form is distinguished by a faulty ectodermal tissue development that includes complete or partial absence of sweat glands and is also known as  Christ-Siemens-Touraine syndrome More Details which affects one to seven individuals per 10,000 live birth. [5]

ED patients have a well-defined characteristic appearance which is well documented. Freire-Maia defined ED [5] as any syndrome that exhibits at least two of the following features: Trichodysplasia (abnormal hairs), abnormal dentition, onchodysplasia (abnormal nails), and dyshidrosis (abnormal or missing sweat glands). Other orofacial characteristic of this syndrome includes anodontia or hypodontia, hypoplastic conical teeth, hypotrichosis, and protuberant lips.

Prosthetic options for management of ED can include fixed, removable, or implant-supported prosthesis; [6] that may be employed individually or in combination to provide an optimal result. [2],[3] Here we report with a case of HAED in a 22-year-old female patient and present the prosthetic management of a young female patient with implant supported fixed partial denture (FPD).


   Case Report Top


A 22-year-old female reported with complaints of multiple missing teeth in upper and lower anterior region. Patient also complained of skin appeared dry with decreased sweating. Patient gave history of difficulty in mastication and swallowing. On examination, patient's skin was dry and soft on face and increased thickness of nails; palm and sole were normal. She is normotensive and febrile with no mental retardation. She did not give positive family history. Her chest and skull radiographs and organ function tests were normal. Intraoral examination showed normal appearing mucosa and salivary output was apparently normal. The patient had 18, 15, 12, 22, 27, 28, 38, 45, and 48 missing teeth with generalized microdontia. The canines had cone shaped clinical crowns, and molars appeared malformed [Figure 1] with obliterated occlusal tables. Permanent maxillary right central incisor was present, which is a rare finding. Vertical dimensions were reduced due to a lack of occlusion and vertical stop causing protuberant lips. The panoramic radiograph confirmed presence of retained primary teeth in both the arches. With the above findings and orthopantomogram (OPG) figure, we came to a conclusion and diagnosed as HAED.
Figure 1: Intraoral photograph

Click here to view


Treatment objective

  • To restore the missing teeth [6]
  • To increase the vertical dimension
  • To correct size of teeth
  • Alignment of teeth. [7]
Treatment plan included compensating microdontia by increasing tooth size using FPD retained by implant. To use implants, enough space was required; so orthodontic alignment of teeth was required. As teeth were distributed unevenly, it had to be aligned to gain space for implants.

Retained deciduous teeth were extracted and orthodontic correction [Figure 2] done with aligning and leveling. Enough space for implant was created within 1 year.
Figure 2: Orthopantomograph during orthodontic correction treatment

Click here to view


Two implants [Figure 3] were placed in region of 12, 13 and one in the region of 45.
Figure 3: Orthopantomograph with implants

Click here to view


Three months were given for osseointegration. In the meantime, crown preparation was done for required teeth for FPD. Wax try-in was done for FPD to restore vertical dimension. Final FPD [Figure 4] was given and orthopantomograph [Figure 5] taken posttreatment.
Figure 4: Intraoral photograph posttreatment

Click here to view
Figure 5: Orthopantomograph posttreatment

Click here to view


The prosthetic rehabilitation provides the patient with esthetic, functional, and phonetic benefits, as well as a better outlook towards her social life and peers oral hygiene instructions, were given to the patient, and he was asked to report for follow up and assessment of her fixed prosthetic appliance after 6 months.


   Discussion Top


The typical treatment of a patient with ED is achieved in several phases, depending on the craniofacial development, thus requiring a comprehensive and multidisciplinary approach. The different treatment modalities are dictated by manifestation of level of hypodontia [8] and resulting malocclusions. Numerous clinical reports have demonstrated importance of prosthetic dental treatment in ED patients for psychological and psychosocial reasons. A number of reviews regarding treatment of ED has showed that implant therapy in early infancy or adolescent age while little information is present in literature for ED patient above 18 years of age. [8] Prosthodontic treatment of ED can include fixed, removable, or implant supported prosthesis of which implant and fixed partial prosthodontics are the most frequent modalities. [9]

In removable prosthesis, frequently the oral mucosa is dry due to a decrease in the number of mucous glands and lesser quantity of saliva which makes retention of removable prosthesis more difficult. Overdentures can be used to restore ideal occlusion and usually allow preservation of existing dentition, but these require rigorous oral hygiene regimes to avoid development of caries and periodontal problems. [10] This case report describes a routine method for fixed prosthodontic [11] treatment of a patient with ED. As the patient was older than 18 years, the growth period was assumed to be over that would have resulted in the failure of FPDs as shown by Hogeboom. [12] A study by Guckes et al., [13],[14] showed that patients younger than 18 years had a hazard ratio of 2.8 compared to the patients older than 18 years. So for young patients, FPDs should be avoided as they could interfere with jaw growth and implants should be recommended and vice versa in patients > 18 years. Excellent oral hygiene is crucial for successful treatment of patients with this condition. The dental appearance of patients affected by ED has to be improved for physiologic and psychologic reasons, so that they can lead a normal social life with self-esteem.


   Conclusion Top


This clinical report demonstrates the implant retained FPD as a suitable treatment while treating an adult patient with ED. This treatment option gives excellent result, which not only improves masticatory function but also enhances esthetics, allowing patients to lead a normal social life.

 
   References Top

1.Thurnam J. Two cases in which skin, hair and teeth were very imperfectly developed. Med Chir Trans 1848;31:71-82.  Back to cited text no. 1
    
2.Weech AA. Hereditary ectodermal dysplasia (congenital ectodermal defect). Am J Dis Child 1929;37:766-9.  Back to cited text no. 2
    
3.Rajendran R, Sivapathasundaram B. Shafer's textbook of oral pathology. 5 th ed. Philadelphia: Saunders; 1983.  Back to cited text no. 3
    
4.Yenisey M, Guler A, Unal U. Orthodontic and prosthodontic treatment of ectodermal dysplasia: A case report. Br Dent J 2004;196:677-9.  Back to cited text no. 4
    
5.Pigno MA, Blackman RB, Cronin RJ, Cavazos E. Prosthodontic management of ectodermal dysplasia: A review of literature. J Prosthet Dent 1996;76:541-5.  Back to cited text no. 5
    
6.NaBadalung DP. Prosthodontic rehabilitation of an anhidrotic ectodermal dysplasia patient: A clinical report. J Prosthet Dent 1999;81:499-502.  Back to cited text no. 6
    
7.Suri S, Carmichael RP, Tompson BD. Simultaneous functional and fixed applied therapy for growth modification and dental alignment prior to prosthetic habilitation in hypohidrotic ectodermal dysplasia: A clinical report. J Prosthet Dent 2004;92:428-33.   Back to cited text no. 7
    
8.Hobkirk JA, Brook AH. The management of patients with severe hypodontia. J Oral Rehabil 1980;7:289-98.  Back to cited text no. 8
    
9.Winstanley RB. Prosthodontic treatment of patients with hypodontia. J Prosthet Dent 1984;52:687-91.   Back to cited text no. 9
    
10.Itthagarun A, King NM. Ectodermal dysplasia: A review and case report. Quintessence Int 1997;28:595-602.   Back to cited text no. 10
    
11.Hickey AJ, Vergo JT. Prosthetic treatments for patients with ectodermal dysplasia. J Prosthet Dent 2001;86:364-8.   Back to cited text no. 11
    
12.Hogeboom FE. Restorative procedures for children with ectodermal dysplasia. ASDC J Dent Child 1961;28:62-72.   Back to cited text no. 12
    
13.Guckes AD, Scurria MS, King TS, McCarthy GR, Brahim JS. Prospective clinical trial of dental implants in persons with ectodermal dysplasia. J Prosthet Dent 2002;88:21-5.  Back to cited text no. 13
    
14.Guckes AD, Brahim JS, McCarthy GR, Rudy SF, Cooper LF. Using endosseous dental implants for patients with ectodermal dysplasia. J Am Dent Assoc 1991;122:59-62.  Back to cited text no. 14
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]


This article has been cited by
1 Clinical outcomes of implant therapy in ectodermal dysplasia patients: a systematic review
Y. Wang,J. He,A.M. Decker,J.C. Hu,D. Zou
International Journal of Oral and Maxillofacial Surgery. 2016;
[Pubmed] | [DOI]



 

Top
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Case Report
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed1527    
    Printed38    
    Emailed1    
    PDF Downloaded77    
    Comments [Add]    
    Cited by others 1    

Recommend this journal