Prosthodontic management of the literature

The University of Texas Health

of ectodermal
J.

dysplasia:

A review

Mark A. Pigno, DDS, Ronald B. Blackman, DDS, MS,b Robert Cronin, Jr., DDS, MS, and Edmund Cavazos, DDSd
Science Center at Sao Antonio, San Antonio,

Tex.

Ectodermal dysplasia is commonly a difficult condition to manage 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. This article reviews the literature that pertains to the prosthodontic treatment of the disorder and the review includes considerations in behavior management and timing of treatment. (J Prosthet Dent 1996;76:541-5.)

ctodermal dysplasia (ED) is a hereditary disorder characterized by abnormal development of certain tissues and structures of ectodermal origin1 Freire-MaiaZ defined the nosologic group of ED as any syndrome that exhibits at least two of the following features: (1) trichodysplasia (abnormal hair), (2) abnormal dentition, (3) onchodysplasia (abnormal nails), and (4) dyshidrosis (abnormal or missing sweat glands). Certain oral and facial characteristics may also be associated with the syndrome. Because there are more than 100 different depend on the ED syndromes, 1.3clinical manifestations specific syndrome afflicting an individual.-* The most frequently reported ED syndrome is X-linked hypohidrotic dysplasia,9 also known as Christ-SiemensTouraine syndrome,7 which affects one to seven individuals per 10,000 live births.7 Orofacial characteristics of this syndrome include anodontia or hypodontia, hypoplastic conical teeth, underdevelopment of the alveolar ridges, frontal bossing, a depressed nasal bridge, protuberant lips, and hypotrichosis.g-12 The characteristics associated with ED will often result in afflicted individuals having an abnormal appearance. Normal social and psychologic development of young ED patients dictates that they look and feel as normal as possible.13-I6 Therefore, dental appearance in these patients is extremely important because it can affect their self-esteem. Numerous clinical reports have demonstrated the importance of prosthetic dental treatment in ED patients for physiologic and psychosocial reasons.16-49 Prostbodontic treatment of ED can include fixed, removable, or implant-supported prostheses.14J6-4g These treatment approaches can be used individually or in comAssistant Professor, Department of Prosthodontics. bAssociate Professor, Department of Prosthodontics. Associate Professor and Head, Graduate Prosthodontics Division, Department of Prosthodontics. dAssociate Professor and Head, Fixed Prosthodontics Division, Department of Prosthodontics.

bination to provide an optimal result. This article will review the literature concerning the prosthodontic treatment of ED, including considerations in patient management and timing of treatment.

METHODS
The literature search for the ED-related articles cited in this review was conducted primarily through the Index to Dental Literature. The inclusion criterion was any English language article that pertained to the prosthodontic management of ED from 1960 to 1995. In addition to the articles obtained from the Index to Dental Literature, the references of all the articles included were reviewed for any previously omitted article that fit the inclusion criterion. Other references cited in this review not directly related to ectodermal dysplasia50-73 were obtained through Medline searches and reference reviews of published articles.

PATIENT ATIONS

MANAGEMENT

CONSIDER-

There is little information in the literature other than clinical reports regarding dental management of the young ED patient. An article by Nowakl* provides the most complete review to date on the dental management of these young patients. Till and Marquesz3 also provide some insights into the subject and are cited in this review. Nowak14 stated that treating the pediatric patient with ED requires the clinician to be knowledgeable in growth and development, behavioral management, techniques in the fabrication of a prosthesis, the modification of existing teeth utilizing composite resins, the ability to motivate the patient and parent in the use of the prosthesis, and the long-term follow-up for the modification and/or replacement of the prosthesis. If the treating dentist is not knowledgeable in one or more of these areas he should obtain consultations or refer treatment when needed. A multidisciplinary team composed of a 541

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pediatric dentist, a prosthodontist, an orthodontist, and an oral and maxillafacial surgeon has been advocated in some reports and recommended to ensure proper treatment of young ED patients.23,44,47 There is not a definitive time to begin treatment, but Till and Marquesz3 recommend that an initial prosthesis be delivered before the child begins school so that the child has a normal appearance and time to adapt to the prosthesis. Ultimately, the decision to begin treatment should be made by the treating dentist along with the parents and patient.* Because individuals with ED are quite young when they are evaluated for treatment, the treating dentist should have some knowledge and ability in the behavioral management of pediatric patients.14 Nussbaum and CarreP advocate sedation for managing problem children who need extensive prosthodontic treatment. Nowak14 does not recommend sedation, stating that it will result in prosthesis failure because ultimate success depends on patient understanding and compliance. Instead of sedation, Nowak advocates conditioning to the pending dental procedures by a tell-show-do53 approach. Other authors have also successfully used this conditioning approach.32,33 According to Nowak,14 a series of introductory visits may be needed before treatment commences, to attain the required patient trust. Parents may facilitate or impede the success of this process, and their involvement must be assessed on an individual basis. When treatment commences, it is important that the patient understand each procedure and its outcome. In addition, the child and parents should have a reasonable idea ofwhat the final prosthesis will look like, how it will benefit the patient, how to care for it, and any limitations that it will have.14 Clinical case studies by Sarnat et a1.34 and Nomura et a1.35 have indicated that jaw growth in individuals with ED is within normal limits. Although these studies are convincing, each study involved only one subject. Studies with larger sample sizes are needed before any definitive conclusions can be made. Regardless ofwhether ED affects jaw growth, it is important that young ED patients be seen regularly after initial prosthesis delivery until their growth is complete, to resolve problems related to this factor.14,35 Common problems are usually related to loss of prosthesis retention or occlusal changes caused by erupting teeth or jaw growth. Nowakl* recommends that the patient and parents be briefed on these types of changes in advance and informed that when growth and development are complete, a more permanent treatment can be rendered.

rigid connectors should be avoided in young, actively growing patients. This is because rigid FPDs could interfere with jaw growth, especially if the prosthesis crosses the midline.l* Hogeboom18 presented a case that dramatically demonstrated the occurrence of jaw growth in an individual treated for ED in which the two segments of a detachable fixed prosthesis separated at the midline because of transverse jaw growth. Individual crown restorations have no age restrictions related to jaw growth, but larger pulp sizes and shorter crown heights may cause concerns4 In spite of these concerns, crowns are often used in the treatment of young ED patients.17-3 Recently, direct composite restorations have become the more desirable method of restoring normal morphology to hypoplastic teeth commonly found in ED patients. 14,23,24 Crowns and direct composite restorations are often used in combination with removable partial dentures (RPDs) in the prosthodontic management of these patients. 18-23They are usually necessary to provide proper contours on the hypoplastic teeth that will be used as abutments for RPDs. Also, orthodontic treatment may be needed to ahgn the teeth into acceptable positions before RPD fabrication.21-24

Removable

prosthodontics

TREATMENT CONSIDERATIONS Fixed prosthodontics

Fixed sively in afflicted addition, are first 542 prosthodontic treatment is seldom used excluthe treatment of ED, primarily because many individuals have a minimal number of teeth. In ED patients are often quite young when they treated, and fixed partial dentures (FPDs) with

Removable prosthodontics is the most frequently reported treatment modality for the dental management of ED.s-44 Because anodontia or hypodontia is typical in individuals with this condition, complete dentures, partial dentures, or overdentures are often part of the treatment provided. Although complete dentures can provide an acceptable esthetic and functional result, underdevelopment of the edentulous alveolar ridges in individuals with ED can compromise denture retention and stability. When there are teeth present for support, overdentures are a desirable treatment option for these patients.14,36-44 Crumso provided an excellent overview of the advantages of conventional overdentures as opposed to complete dentures. One important advantage is that overdentures preserve alveolar bone. Van Waas et a1.51 verified this claim with a well-designed, randomized, controlled clinical trial. The trial compared average mandibular bone reduction in 74 patients treated randomly with either an immediate overdenture on two mandibular canines or an immediate complete denture. There was a significant reduction in alveolar bone loss in the overdenture patients after 2 years. Preservation of alveolar bone is imperative in individuals with ED, because they must depend on the alveolar ridges for prosthesis support from an early age. If an overdenture is fabricated, retention can be augmented by various attachments anchored to the available teeth.52 When there are no teeth available for complete denture support, vestibuloplasty and ridge augmentation have been mentioned as treatment options that may enhance the prosthodontic management of ED.14.24
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As for accommodation by young ED patients to complete dentures, published cases usually report good adaccommodaaptation to the prostheses. 25-44To facilitate tion in young children, some clinicians have reported the delivery of one denture at a time.25,30,33J8 Till and Marquess3 advocate this method and recommend that the denture made for the arch with the best prognosis be delivered first, followed by delivery of the second denture 2 to 4 months later. An important factor in patient acceptance is the appearance of the denture teeth in rielation to the patients age. 14sz4 The dentition stage depicted in the denture should be appropriate. This is accomplished by using primary prosthetic teeth for the very young patients and making modifications to existing or new dentures as the patient grows older. Periodic recalls of young ED patients are also important because prosthesis modification or replacement will be needed as a result of continuing growth and development. 14,24,34,35 In addition to adjustments related to fit, the occlusion of a prosthesis must be monitored for changes because of jaw growth.35 Other problems related to removable prostheses are speech difficulties, dietary limitations, and loss of the prosthesis.14

Implant

prosthodontics

The literature indicates that endosteal implants are being used more widely in the prosthodontic management of ED.45-4g Guckes et a1.46 have reported preliminarily a 90% dental implant success rate at second-stage surgery in ED patients 13 to 69 years old, as part of an ongoing clinical trial at the National Institutes of Health. This success rate approaches the 95% success rate reported for the non-ED patients in the study. The specifics regarding the study design were not provided; the only problem mentioned in the preliminary report was labial angulation of the implants because of alveolar ridge anatomy. Ekstrand and Thomsson,45 Bergendal et a1.,47 Smith et a1.,48 and Cronin et a14 have also reported situations in which endosteal implants were successfully used in the prosthodontic management of ED. A number of studies indicate an improvement in the physiologic and psychosocial function of adult patients with an implant-supported denture when compared with their condition before implants were placed or to an edentulous control group with complete dentures.55-62 Kent? provides a review of the effects of dental implants on psychologic and social well-being. He found flaws with some of the studies that were retrospective as opposed to prospective, because the retrospective studies did not include a control group or did not use validated questionnaires. In spite of the flaws, he concludes that there is a consistent and clinically significant pattern of results to support the claim that dental implants can positively affect well-being and quality of life.61 As already noted, prosthodontic treatment is extremely important in ED patients for physiologic and psychosocial reasons.13-16 As
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a result, implant-supported prostheses seem to offer a desirable treatment option for these patients. Another reason to consider dental implants in the treatment of ED is the beneficial impact they could possibly have on the preservation of alveolar bone.63-66 AS previously mentioned, the alveolar ridges of individuals with ED are underdeveloped because of the lack of tooth development. These alveolar ridges must support a dental prosthesis over the course of a lifetime. Thus, treatment measures that will maintain alveolar bone and enhance the prognosis for future prosthodontic treatment are extremely important, especially in individuals with total anodontia. Although the concept of preventive implantation with endosteal dental implants has been advanced in the literature,63-66 there are no studies that demonstrate its effectiveness in maintaining alveolar bone when compared with a control group. More research is needed in this area before the treatment concept can be universally advocated. Recently concern has been expressed in the literature regarding placement of osseointegrated implants in a growing jaw. 46,4g,67-73 Guckes et a1.46 discuss this issue and recommend that implant placement be postponed until age 13 because of possible implant movement caused by jaw growth, the expense of frequent remakes, and the lack of clinical experience in placing implants in young children. A 1989 Consensus Conference on Implantology67 concluded that implants should not be placed until maximum jaw growth has occurred, which was reported as up to 15 years of age. In an editorial, Lekholme8 concurs with criteria concerning maximum jaw growth, giving age guidelines of 14 to 15 years of age for girls and a year later for boys. He also recommends that an individuals growth curve be studied before any implant placement procedure is started. Studies carried out by Odman et a1.,6g Thilander et a1.,70 and Sennerby et al.71 demonstrated that implants placed in the jaws of growing pigs do not behave like normal teeth but instead become ankylosed in the bone. The sample size of five pigs used for the studies was small and six of the 20 implants placed were lost. Despite the problems, the results obtained from the remaining 14 implants clearly show that dental implants placed in the jaws of growing pigs become ankylosed. It can be assumed that implants placed in children would behave similarly. Odman et a1.6g concluded that implants should not be placed in children until the permanent dentition is fully erupted. Thilander et a170 concurred with this finding but did not rule out placement of implants anterior to the canines in some children with total or partial anodontia, for psychosocial and functional reasons. Oesterle et alT2 discuss maxillary jaw growth and the possible consequences of early implant placement in the maxillae. They stress that implants placed in postpubertal or postgrowth patients have a greater likelihood of success. In a similar article, Cronin et al.@ provide a descrip543

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tion of mandibular jaw growth and the possible consequences of early implant placement in the mandible. They concluded that implants placed after age 15 years for girls and 18 years for boys provided the most predictable prognosis. They also cautioned against the overgeneralization of clinical reports, because of the variation in growth among individuals, and recommended retrievability in implant-supported prostheses fabricated for children to facilitate adjustments related to growth and development. According to Cronin et a1.4gand Oesterle et a1.,72possible consequences of early implant placement include implant submergence because of jaw growth, implant exposure because of bone resorption associated with jaw growth, implant movement because of jaw growth, and limitation of jaw growth if the implants. are connected by a rigid prosthesis that crosses the midline. Finally, Thilander et a1.73longitudinally followed 27 single-crown restored implants placed in 15 adolescents whose ages ranged from 13 to 19 years. All of the subjects studied had fully erupted permanent teeth with the exception of third molars. Restoration position was monitored with standardized radiography and measurements from stone casts. The position was assessed for changes after a 3-year period. Although the sample size was small and implant location was not standardized, there was a positive correlation between craniofacial growth and infraocclusion of the restorations. Even though minor infraocclusion did occur in most of the restorations, they concluded that implants are acceptable for that age group provided that growth and development is complete, with all teeth fully erupted. The authors stress that the dental and skeletal maturity, and not the chronological age of the patient is important to avoid infraocclusion of an implant restoration.73 It is clear from the literature that the timing of treatment is important if implants are placed in young ED patients because of possible complications resulting from jaw growth. SUMMARY The prosthodontic management of ED requires a broad knowledge base to handle the special problems associated with treatment. For this reason, a multidisciplinary team approach is recommended for optimal dental management of the condition. Although it is important to provide early treatment, it must be remembered that any prosthesis made for a young patient must be closely monitored for needed adjustments or for a replacement prosthesis made necessary by growth and development. A 6- to 12-month recall schedule until skeletal growth is complete is advised. Also, a tell-show-do approach to treatment is recommended for the young ED patient. Treatment can involve fixed, removable, or implant prosthodontics, singly or in combination. Regarding fmed prosthodontics for dental management of ED, FPDs should be avoided in young, actively growing patients because they could interfere with jaw
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growth. This is especially true if the prosthesis crosses the midline. As for the hypoplastic teeth common with ED, direct composites or crowns are often needed to restore proper contours to the teeth. Removable prosthodontics is the most frequent treatment modality used for dental management of ED. Although complete dentures are an acceptable form of treatment, overdentures or RPDs supported by natural teeth are desirable for preservation of alveolar bone. When removable prostheses are fabricated for ED patients, the dentition stage depicted should be appropriate. In addition, it may be necessary to fabricate and deliver one denture at a time in young patients with no denture experience, to facilitate accommodation. Implant-supported restorations can improve physiologic and psychosocial function when compared with complete dentures, but their placement in growing jaws can cause complications. When implant placement in young ED patients is being considered, their dental and skeletal maturity, not their chronological age, should be the determining factor. An individuals growth curve can help in this determination. For dental implants to become a more desirable and recommended treatment option for young ED patients, more research is needed to determine guidelines for dental implant placement in children and the effectiveness of the concept of preventive implantation with dental implants in maintaining alveolar bone.
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