Oral Rehabilitation of a Patient with Ectodermal Dysplasia with Prosthodontics Treatment
Identifieur interne : 001C93 ( Pmc/Corpus ); précédent : 001C92; suivant : 001C94Oral Rehabilitation of a Patient with Ectodermal Dysplasia with Prosthodontics Treatment
Auteurs : Srinivas NallanchakravaSource :
- Indian Journal of Dermatology [ 0019-5154 ] ; 2013.
Abstract
Ectodermal dysplasia (ED) is a rare, congenital disease that involves the sweat glands, scalp hair, nails, skin pigmentation, and craniofacial structure. Oral symptoms of ED include multiple tooth abnormalities (such as hypodontia, anadontia, impacted teeth, and peg-shaped or conical anterior teeth) and lack of normal alveolar ridge development. A 12-year-old male patient, in the absence of any other systemic abnormalities, exhibited typical characteristics of ED, visited our department of pedodontics and preventive dentistry at Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre. In the clinical and radiographic evaluation, it was occurred that he had only maxillary and mandibular first molars in his mouth. A maxillary and mandibular denture with clasps for retention was fabricated for prosthodontic rehabilitation after considering his growth and the number and condition of his present teeth. At the 18-month follow-up no major complications occurred and further future treatment included implants retained fixed partial dentures.
Url:
DOI: 10.4103/0019-5154.110851
PubMed: 23723492
PubMed Central: 3667304
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<front><div type="abstract" xml:lang="en"><p>Ectodermal dysplasia (ED) is a rare, congenital disease that involves the sweat glands, scalp hair, nails, skin pigmentation, and craniofacial structure. Oral symptoms of ED include multiple tooth abnormalities (such as hypodontia, anadontia, impacted teeth, and peg-shaped or conical anterior teeth) and lack of normal alveolar ridge development. A 12-year-old male patient, in the absence of any other systemic abnormalities, exhibited typical characteristics of ED, visited our department of pedodontics and preventive dentistry at Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre. In the clinical and radiographic evaluation, it was occurred that he had only maxillary and mandibular first molars in his mouth. A maxillary and mandibular denture with clasps for retention was fabricated for prosthodontic rehabilitation after considering his growth and the number and condition of his present teeth. At the 18-month follow-up no major complications occurred and further future treatment included implants retained fixed partial dentures.</p>
</div>
</front>
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<author><name sortKey="Abson, Kg" uniqKey="Abson K">KG Abson</name>
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<author><name sortKey="Brahim, Js" uniqKey="Brahim J">JS Brahim</name>
</author>
<author><name sortKey="Mccarthy, Gr" uniqKey="Mccarthy G">GR McCarthy</name>
</author>
<author><name sortKey="Rudy, Sf" uniqKey="Rudy S">SF Rudy</name>
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<pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Indian J Dermatol</journal-id>
<journal-id journal-id-type="iso-abbrev">Indian J Dermatol</journal-id>
<journal-id journal-id-type="publisher-id">IJD</journal-id>
<journal-title-group><journal-title>Indian Journal of Dermatology</journal-title>
</journal-title-group>
<issn pub-type="ppub">0019-5154</issn>
<issn pub-type="epub">1998-3611</issn>
<publisher><publisher-name>Medknow Publications & Media Pvt Ltd</publisher-name>
<publisher-loc>India</publisher-loc>
</publisher>
</journal-meta>
<article-meta><article-id pub-id-type="pmid">23723492</article-id>
<article-id pub-id-type="pmc">3667304</article-id>
<article-id pub-id-type="publisher-id">IJD-58-241c</article-id>
<article-id pub-id-type="doi">10.4103/0019-5154.110851</article-id>
<article-categories><subj-group subj-group-type="heading"><subject>E-Case Report</subject>
</subj-group>
</article-categories>
<title-group><article-title>Oral Rehabilitation of a Patient with Ectodermal Dysplasia with Prosthodontics Treatment</article-title>
</title-group>
<contrib-group><contrib contrib-type="author"><name><surname>Nallanchakrava</surname>
<given-names>Srinivas</given-names>
</name>
<xref ref-type="aff" rid="aff1"></xref>
<xref ref-type="corresp" rid="cor1"></xref>
</contrib>
</contrib-group>
<aff id="aff1"><italic>From the Department of Pedodontics and Preventive Dentistry, Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre, Dilshuknagar, Hyderabad, Andhra Pradesh, India</italic>
</aff>
<author-notes><corresp id="cor1"><italic><bold>Address for correspondence:</bold>
Dr. Srinivas Nallanchakrava, Department of Pedodontics and Preventive Dentistry, Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre, Dilshuknagar, Hyderabad - 500 060, Andhra Pradesh, India. E-mail: <email xlink:href="cnudent@gmail.com">cnudent@gmail.com</email>
</italic>
</corresp>
</author-notes>
<pub-date pub-type="ppub"><season>May-Jun</season>
<year>2013</year>
</pub-date>
<volume>58</volume>
<issue>3</issue>
<fpage seq="c">241</fpage>
<lpage>241</lpage>
<history><date date-type="received"><month>1</month>
<year>2011</year>
</date>
<date date-type="accepted"><month>8</month>
<year>2011</year>
</date>
</history>
<permissions><copyright-statement>Copyright: © Indian Journal of Dermatology</copyright-statement>
<copyright-year>2013</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-sa/3.0"><license-p>This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
</license>
</permissions>
<abstract><p>Ectodermal dysplasia (ED) is a rare, congenital disease that involves the sweat glands, scalp hair, nails, skin pigmentation, and craniofacial structure. Oral symptoms of ED include multiple tooth abnormalities (such as hypodontia, anadontia, impacted teeth, and peg-shaped or conical anterior teeth) and lack of normal alveolar ridge development. A 12-year-old male patient, in the absence of any other systemic abnormalities, exhibited typical characteristics of ED, visited our department of pedodontics and preventive dentistry at Panineeya Mahavidyalaya Institute of Dental Sciences and Research Centre. In the clinical and radiographic evaluation, it was occurred that he had only maxillary and mandibular first molars in his mouth. A maxillary and mandibular denture with clasps for retention was fabricated for prosthodontic rehabilitation after considering his growth and the number and condition of his present teeth. At the 18-month follow-up no major complications occurred and further future treatment included implants retained fixed partial dentures.</p>
</abstract>
<kwd-group><kwd><italic>Ectodermal dysplasia</italic>
</kwd>
<kwd><italic>fixed partial dentures</italic>
</kwd>
<kwd><italic>hypodontia</italic>
</kwd>
<kwd><italic>implants</italic>
</kwd>
<kwd><italic>overdentures</italic>
</kwd>
<kwd><italic>prosthodontic treatment</italic>
</kwd>
</kwd-group>
</article-meta>
</front>
<body><sec id="sec1-1"><title>Introduction</title>
<boxed-text position="float"><p><bold>What was known?</bold>
</p>
<p>Ectodermal dysplasia is a rare, congenital disease that involves the sweat glands, scalp hair, nails, skin pigmentation, and craniofacial structure with oral symptoms like hypodontia, anadontia, etc.</p>
</boxed-text>
<p>Ectodermal dysplasia (ED) have been described as a group of disorders of morphogenesis displaying two or more of the symptoms of trichodysplasia, dental anomalies, onychodysplasia, and dyshidrosis.[<xref ref-type="bibr" rid="ref1">1</xref>
] It is usually described as being hypohidrotic or hidrotic, depending upon the degree of sweat gland function. Congenital malformation of teeth, hair, nails, or sweat glands may occur either as single isolated malformations or as a part of an ectodermal dysplasia syndrome. Anhidrotic ectodermal dysplasia is considered to be a triad of hypodontia or anodontia, hypotrichosis, and hypohidrosis, and associated with other components that result from defective development of structures of ectodermal origin.[<xref ref-type="bibr" rid="ref2">2</xref>
] Anhidrotic ectodermal dysplasia is X-linked condition, and is found in all races, with an incidence of 1-7 per 100.000 live births.[<xref ref-type="bibr" rid="ref3">3</xref>
] Affected males usually have prominent supraorbital ridges, frontal swelling, thin eyebrow, fine-linear wrinkles, sparse hair, detective nails, and saddle nose.[<xref ref-type="bibr" rid="ref3">3</xref>
<xref ref-type="bibr" rid="ref4">4</xref>
] Due to the altered anatomy in the lower third of the face, they resemble to edentulous old individuals.</p>
<p>Oral characteristics include complete or partial hypodontia, anodontia, impacted teeth, loss of vertical dimensions of occlusion, protuberant lips, malformed and peg-shaped or conical teeth, and lack of alveolar growth. Oral symptoms of ED include multiple tooth abnormalities and lack of normal alveolar ridge development can complicate the restoration of an ideal occlusion, especially in a growing child. Lack of alveolar growth frequently results in increased interocclusal distance, which allows optimum artificial tooth placement.[<xref ref-type="bibr" rid="ref5">5</xref>
] For rehabilitation, it is crucial to know the age, number and condition of present teeth, and the state of growth of the patient.</p>
</sec>
<sec id="sec1-2"><title>Case Report</title>
<p>A 12-year-old boy reported to our out patient department with a chief complaint of missing teeth, inability to eat, difficulty in speech. The patient even had a psychological set back due to lack of esthetics. The general medical and family history was non contributory. The boy presented with classical features of ectodermal dysplasia: Partial anodontia [Figures <xref ref-type="fig" rid="F1">1</xref>
and <xref ref-type="fig" rid="F2">2</xref>
], hypohidrosis, hypotrichosis, frontal bossing, saddle nose, diminished lower facial height, sparse scalp hair, missing eye lashes and eyebrows, protuberant lips. The intraoral examination revealed presence of maxillary and mandibular first molars [<xref ref-type="fig" rid="F2">Figure 2</xref>
]. The alveolar ridge was efficient in both height and width. The oral mucosa was dry in appearance. This was the patient's first dental visit and he never wore any dentures before.</p>
<fig id="F1" position="float"><label>Figure 1</label>
<caption><p>Intra oral view of only maxillary and mandibular molars in occlusion</p>
</caption>
<graphic xlink:href="IJD-58-241c-g001"></graphic>
</fig>
<fig id="F2" position="float"><label>Figure 2</label>
<caption><p>Frontal view of the face</p>
</caption>
<graphic xlink:href="IJD-58-241c-g002"></graphic>
</fig>
<sec id="sec2-1"><title>Current treatment done and future treatment plans</title>
<p>Removable prosthesis through dentures was planned for maxilla and mandible. Steps included are primary impression followed by special tray fabrication [<xref ref-type="fig" rid="F3">Figure 3</xref>
], then border molding was done on special trays and dual impression technique opted for better impression recording [Figures <xref ref-type="fig" rid="F4">4</xref>
and <xref ref-type="fig" rid="F5">5</xref>
]. Then wax rims were made and jaw relationship is recorded, followed by trial dentures and finally finished partial dentures [<xref ref-type="fig" rid="F6">Figure 6</xref>
] were given to complete oral and psychological rehabilitation [<xref ref-type="fig" rid="F7">Figure 7</xref>
]. Future treatment includes implants and fixed partial dentures for both maxilla and mandible.</p>
<fig id="F3" position="float"><label>Figure 3</label>
<caption><p>Special trays for maxilla and mandible</p>
</caption>
<graphic xlink:href="IJD-58-241c-g003"></graphic>
</fig>
<fig id="F4" position="float"><label>Figure 4</label>
<caption><p>Secondary dual impression for maxilla</p>
</caption>
<graphic xlink:href="IJD-58-241c-g004"></graphic>
</fig>
<fig id="F5" position="float"><label>Figure 5</label>
<caption><p>Secondary dual impression for mandible</p>
</caption>
<graphic xlink:href="IJD-58-241c-g005"></graphic>
</fig>
<fig id="F6" position="float"><label>Figure 6</label>
<caption><p>Final dentures for maxilla and mandible</p>
</caption>
<graphic xlink:href="IJD-58-241c-g006"></graphic>
</fig>
<fig id="F7" position="float"><label>Figure 7</label>
<caption><p>Oral and psychologically rehabilitation done</p>
</caption>
<graphic xlink:href="IJD-58-241c-g007"></graphic>
</fig>
</sec>
</sec>
<sec sec-type="discussion" id="sec1-3"><title>Discussion</title>
<p>Hypodontia and anodontia were frequently seen in many cases of ectodermal dysplasia.[<xref ref-type="bibr" rid="ref6">6</xref>
<xref ref-type="bibr" rid="ref7">7</xref>
] Prosthetic rehabilitation of patients with ED is an ordinary process. Early and extensive dental treatment is needed throughout childhood because of the absence of most of the deciduous and permanent dentition. A multidisciplinary team approach to management of these patients is recommended. [<xref ref-type="bibr" rid="ref8">8</xref>
] Osseo-integrated implants should be an alternative treatment in older patients with ED.[<xref ref-type="bibr" rid="ref9">9</xref>
–<xref ref-type="bibr" rid="ref13">13</xref>
] Considering the poor economical situation of the present case, it was deemed better to postpone Osseo-integrated implants. This is commonly a difficult condition to manage the patients with prosthodontics because of the typical oral deficiencies and because the afflicted individuals are quite young when they are evaluated for treatment. It is important that these individuals receive dental treatment at an early age for physiologic and psychosocial reasons. Adaptation to the prosthesis depends on various factors as, capability of the specialist, age of the patient, and cooperation in a growing child. A removable partial denture or an overdenture is often a suitable treatment choice, because of the need to easily modify the intraoral prosthesis during rapid growth period[<xref ref-type="bibr" rid="ref12">12</xref>
<xref ref-type="bibr" rid="ref13">13</xref>
] and also these treatment options are easy, affordable, and reversible rehabilitation methods.</p>
<p>The treatment goals for this patient were to establish a functional occlusion with prosthetic rehabilitation and, to obtain an esthetic smile with adult-sized teeth, because any prosthesis was not worn by the patient before. Also nutrition of the patient is improved, and a self-confident appearance is provided.</p>
<boxed-text position="float"><p><bold>What is new?</bold>
</p>
<p>Early and extensive dental treatment is needed throughout childhood. A multidisciplinary approach to management of these patients is recommended.</p>
</boxed-text>
</sec>
</body>
<back><ack><title>Acknowledgement</title>
<p>I acknowledge the staff of Department of Prosothodontics.</p>
</ack>
<fn-group><fn fn-type="supported-by"><p><bold>Source of Support:</bold>
Nil</p>
</fn>
<fn fn-type="conflict"><p><bold>Conflict of Interest:</bold>
Nil.</p>
</fn>
</fn-group>
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