- Email: [email protected]
Hypodontia due to ectodermal dysplasia: Rehabilitation with very early dental implants
Available online 23 April 2013
Available online at
www.sciencedirect.com
Case report Hypodontie d’une dysplasie ectodermique : traitement par implantologie tre`s pre´coce C. Paulus*, P. Martin Service de chirurgie maxillo-faciale, hoˆpital Femme–Me`re-Enfant, 59, boulevard Pinel, 69500
Hypodontia due to ectodermal dysplasia: Rehabilitation with very early dental implants
Bron, France
Summary Introduction. Anhidrotic ectodermal dysplasia is a rare disease combining hypodontia, hypotrichosis, and hypohidrosis. The quality of life is greatly impaired very early, with major difficulty for feeding and the drawbacks of usual dental prostheses. Early implant placement is a therapeutic alternative. It is usually performed in areas of stable growth, such as the mandibular symphysis. We report the case of very early implant placement in a child presenting with hypodontia related to ectodermal dysplasia. Case presentation. A 6-year-old male patient was treated with maxillary and mandibular implant-borne prosthetic rehabilitation. Five implants were placed in the mandible and seven in the maxilla. The esthetic and functional outcome was satisfactory, improving the quality of life. Discussion. Very early implant-borne prosthetic rehabilitation is an alternative, which could become a first line treatment. It restores orofacial functions allowing for a better development of maxillo-facial bones. This alternative is not without risks. But is it acceptable to wait until teenage with an inadequate removable prosthesis, because growth is not completed? ß 2013 Elsevier Masson SAS. All rights reserved. Keywords: Implant, Hypodontia, Ectodermal dysplasia
Re´sume´ Introduction. La dysplasie ectodermique anhydrotique est une maladie rare associant hypodontie, hypotrichose et hypohydrose. Leur qualite´ de vie est conside´rablement alte´re´e de`s le plus jeune aˆge, notamment par les difficulte´s alimentaires lie´es aux inconve´nients des re´habilitations occluso-prothe´tiques conventionnelles. L’implantologie pre´coce est une alternative the´rapeutique. Elle se fait classiquement dans les zones ou` la croissance est stabilise´e comme la re´gion symphysaire mandibulaire. Nous rapportons un exemple d’implantologie tre`s pre´coce chez un enfant atteint d’hypodontie s’inte´grant dans une dysplasie ectodermique. Observation. Un enfant aˆge´ de six ans a be´ne´ficie´ d’une re´habilitation prothe´tique fixe implanto-porte´e maxillaire et mandibulaire. Douze implants ont e´te´ pose´s : cinq mandibulaires et sept maxillaires. Les re´sultats esthe´tiques et fonctionnels ont e´te´ bons, ame´liorant la qualite´ de vie. Discussion. La re´habilitation implanto-porte´e tre`s pre´coce est possible, voire souhaitable, lorsque les conditions sont re´unies chez ces patients. Elle re´tablit des fonctions oro-faciales qui favorisent le de´veloppement des bases squelettiques maxillo-faciales. Il existe des risques lie´s a` cette option the´rapeutique. Mais peut-on attendre jusqu’a` l’adolescence avec une prothe`se amovible non adapte´e, sous pre´texte d’une croissance non acheve´e ? ß 2013 Elsevier Masson SAS. Tous droits re´serve´s. Mots cle´s : Implant, Hypodontie, Dysplasie ectodermique
Introduction DOI of original article: http://dx.doi.org/10.1016/j.revsto.2013.01.009 * Corresponding author. E-mail addresses: [email protected], [email protected]
Ectodermal dysplasia includes a heterogeneous group of rare genetic diseases characterized by development abnormalities of ectodermal structures [1–7]. Anhidrotic ectodermal
2213-6533/$ - see front matter ß 2013 Elsevier Masson SAS. All rights reserved. http://dx.doi.org/10.1016/j.revsto.2013.03.008 Rev Stomatol Chir Maxillofac Chir Oral 2013;114:e5-e8
e5
C. Paulus, P. Martin
dysplasia is a rare disease combining hypodontia, hypotrichosis, and hypohidrosis. Currently, around environ 200 kinds of ectodermal dysplasia, different genetically and clinically have been identified. Anhidrotic ectodermal dysplasia is the most frequent of these ectodermal dysplasias (7/10,000 births). The patient’s quality of life is greatly impaired and this at a very early age. In most of cases, there are abnormalities on the number (anodontia, hypodontia) and shape (conical) of teeth. This often causes major oro-facial dysfunctions and social handicap. Usual dental prostheses are not adapted to a very young patient. Very early implant-borne prosthetic rehabilitation could be an alternative. We report the case of very early implant placement in a child presenting with hypodontia related to ectodermal dysplasia.
Rev Stomatol Chir Maxillofac Chir Oral 2013;114:e5-e8
Figure 2. Panoramic X-ray of two dystrophic germs, 21 and 11.
Observation A 23-month old child was brought to the pediatric stomatology and maxillo-facial surgery unit in January 2005, for ‘‘missing teeth’’. Patient history documented episodes of hyperthermia. An extra-oral examination allowed observing, thin and dry skin with periorbital and peri-buccal wrinkles, periorbital hyperpigmentation, thin and sparse hair fins, a bilateral labial commissural perleche, and a labial vertical height defect (fig. 1). The oral examination revealed three cone shaped canines (23, 33, and 43). The radiological assessment (panoramic X-ray, frontal and lateral teleradiography) allowed visualizing two dystrophic germs (21 and 11) (figs. 2 and 3). The diagnosis of ectodermal dysplasia was made on these findings. Dental rehabilitation was undertaken with removable dental devices. But the lack of stability required multiple adaptations
Figure 1. Thin and dry skin with periorbital and peri-buccal wrinkles, periorbital hyperpigmentation, thin and sparse hair fins, a bilateral labial commissural perleche, and a labial vertical height defect.
e6
Figure 3. Profile teleradiography revealing severe maxillo-mandibular bone resorption.
and several new prostheses (years 2006, 2007, and 2008). The onset of oral candidiasis contributed to the patient’s rejection of removable dental devices. In December 2008, when the child was 5 years and 9 months of age, it was decided to use maxillo-mandibular implantborne prosthetic rehabilitation, after obtaining the parents’ enlightened consent concerning the risks of this therapeutic strategy: multiple general anesthesia (GA) procedures and risk of graft or implant failure. The first GA took place in June 2009, when the child was 6 years old. Teeth 33 and 43 were extracted. Five implants, 3.4 mm in diameter and 13 mm long (Progress, TekkaW, Lyon, France), were placed in the site of teeth 34 to 44, after performing mucoperiosteal flaps. An autogenous bone graft was placed on the lingual and vestibular side of the symphysis. Tooth 13 and impacted 21 and 11 were extracted on the maxilla, after performing a crestal incision, posterior discharge incision, and mucoperiosteal debridement. Seven implants were placed:
Hypodontia due to ectodermal dysplasia
Figure 4. Final results after complete rehabilitation with significant esthetic improvement.
three with a diameter of 4 mm and 13 mm long (in tooth area 13, 14, and 16); three with a diameter of 3.4 mm and 13 mm long (in tooth area 23, 24, and 26); one with a diameter of 3.4 mm 10 mm long in tooth area 11.
Grafts associating autogenous bone and bank bone (OsteoPureW) were placed at the bilateral intra-sinus, vestibular, palatine, and peri-implant level. The wound was closed in two planes with Vicryl 3/0 (EthiconW) after sub-periosteal dissection and striation of the periosteum. Mandibular implants were exposed after 4 months, and transgingival screws were placed during a second GA. Multi-cone pillars were then inserted, as well as a trans-screwed beam. A Bra˚nemarkW type bridge was screwed. The removable superior prosthesis was fractured several times before placement of a fixed maxillary prosthesis, because of chewing on the fixed inferior prosthesis. The maxillary implants were exposed after 20 months, and trans-gingival screws were placed during a third GA. The print and the vertical dimension were evaluated during this third procedure. The final maxillary prosthetic rehabilitation was made with two trans-screwed ceramic and metallic hemi-bridges, freeing the palatine suture. The child immediately found the prostheses to be comfortable, esthetic, and especially efficient. Dental hygiene was significantly improved, and 2 months before the end of rehabilitation, the weight gain had reached 3 kgs, thanks to a rehabilitated chewing function. The phonation was normal; there was no lingual interposition, or infantile deglutition. Concerning the esthetic aspect, the labial ratios were normalized and the vertical deficiency resolved. The perleche and the peri-buccal wrinkles disappeared (fig. 4). The child became more calm and self-confident. His schooling returned to normal and he no longer had any relational problems.
Figure 5. Results after two postoperative months a (panoramic) and b (profile).
The regular radiological follow-up revealed the stability of fixtures and a normal growth of the maxilla (fig. 5). None implant was lost.
Discussion The implementation of intra-osseous implants is often postponed until the end of maxillary and mandibular growth. Some authors have proposed an earlier implantation [1,3,6]. We decide to implement an early implantation, despite our patient’s age (6 years), because of a severe hypodontia severe preventing the use of stable removable prostheses. This had for consequences a great physiological, esthetic, and social impact. The major risks were: short-, medium-, or long-term implant failure; and repeated GAs. But the benefits seemed significantly superior to the risks: early implant placement allowed soliciting maxillo-facial growth thanks to rehabilitated muscular activity;
e7
C. Paulus, P. Martin
social integration was available thanks to esthetic, functional, and psychological improvement. These benefits had been confirmed by other authors [4,7,8].
The maxilla of children with ectodermal dysplasia presents atrophy and bone resorption related to severe dental agenesis [2,9]; it is not totally functional and not adapted to a removable prosthesis. There are no studies on early maxillary implantation. We suppose that implants act as ‘‘external fixtures’’ and stimulate the facial growth by rehabilitating oro-facial functions. Given the very young age, a regular radiological follow-up must be implemented to assess the growth and outcome of implants and peri-implant tissues. Prostheses on early implants should be considered as temporary. This type of treatment should be validated by future studies and long-term results assessed. The issue raised in this case is: ‘‘Should a patient have to endure non-adapted prostheses during his childhood and teenage, because growth is not finished?’’ One of the most interesting perspectives for patients presenting with hypodontia or anodontia is using pluripotent stem cells to induce a regeneration of dental organs [10].
Rev Stomatol Chir Maxillofac Chir Oral 2013;114:e5-e8
References [1]
[2]
[3]
[4]
[5]
[6]
[7]
[8]
[9]
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
e8
[10]
Bjork A, Skieller V. Growth of the maxilla in three dimensions as revealed radiographically by the implant method. Br J Orthod 1977;4:53–64. Bonin B, Saffarzadeh A, Picard A, Levy P, Romieux G, Goga D. Traitement implantaire pre´coce d’un enfant atteint d’une dysplasie ectodermique anhydrotique. A` propos d’un cas. Rev Stomatol Chir Maxillofac 2001;102:313–8. Davarpanah M, Moon JW, Yang LR, Celletti R, Martinez H. Dental implants in the oral rehabilitation of a teenager with hypohidrotic ectodermal dysplasia: report of a case. Int J Oral Maxillofac Implants 1997;12:252–8. Fotso J, Hugentobler M, Kiliaridis S, Richter M. Dysplasie ectodermique anhydrotique. Re´habilitation. Rev Stomatol Chir Maxillofac 2009;110:50–4. Pinheiro M, Freire-Maia N. Ectodermal dysplasias: a clinical classification and a causal review. Am J Med Genet 1994;53: 153–62. Guckes AD, McCarthy 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. 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. Kramer FJ, Baethge C, Tschernitscheck H. Implants in children with ectodermal dysplasia: a case report and literature review. Clin Oral Implants Res 2007;18:140–6. Soubeyrand E, Nicolas J, Labbe´ D, Riscala S, Olive L, Compe`re JF. La dysplasie ectodermique anhydrotique : pre´sentation de quatre observations. Rev Stomatol Chir Maxillofac 2005;106: 328–33. Magloire H, Couble ML. L’implant dentaire biologique : mythe ou re´alite´ ? Rev Stomatol Chir Maxillofac 2011;112:240–8.
Available online at
www.sciencedirect.com
Case report Hypodontie d’une dysplasie ectodermique : traitement par implantologie tre`s pre´coce C. Paulus*, P. Martin Service de chirurgie maxillo-faciale, hoˆpital Femme–Me`re-Enfant, 59, boulevard Pinel, 69500
Hypodontia due to ectodermal dysplasia: Rehabilitation with very early dental implants
Bron, France
Summary Introduction. Anhidrotic ectodermal dysplasia is a rare disease combining hypodontia, hypotrichosis, and hypohidrosis. The quality of life is greatly impaired very early, with major difficulty for feeding and the drawbacks of usual dental prostheses. Early implant placement is a therapeutic alternative. It is usually performed in areas of stable growth, such as the mandibular symphysis. We report the case of very early implant placement in a child presenting with hypodontia related to ectodermal dysplasia. Case presentation. A 6-year-old male patient was treated with maxillary and mandibular implant-borne prosthetic rehabilitation. Five implants were placed in the mandible and seven in the maxilla. The esthetic and functional outcome was satisfactory, improving the quality of life. Discussion. Very early implant-borne prosthetic rehabilitation is an alternative, which could become a first line treatment. It restores orofacial functions allowing for a better development of maxillo-facial bones. This alternative is not without risks. But is it acceptable to wait until teenage with an inadequate removable prosthesis, because growth is not completed? ß 2013 Elsevier Masson SAS. All rights reserved. Keywords: Implant, Hypodontia, Ectodermal dysplasia
Re´sume´ Introduction. La dysplasie ectodermique anhydrotique est une maladie rare associant hypodontie, hypotrichose et hypohydrose. Leur qualite´ de vie est conside´rablement alte´re´e de`s le plus jeune aˆge, notamment par les difficulte´s alimentaires lie´es aux inconve´nients des re´habilitations occluso-prothe´tiques conventionnelles. L’implantologie pre´coce est une alternative the´rapeutique. Elle se fait classiquement dans les zones ou` la croissance est stabilise´e comme la re´gion symphysaire mandibulaire. Nous rapportons un exemple d’implantologie tre`s pre´coce chez un enfant atteint d’hypodontie s’inte´grant dans une dysplasie ectodermique. Observation. Un enfant aˆge´ de six ans a be´ne´ficie´ d’une re´habilitation prothe´tique fixe implanto-porte´e maxillaire et mandibulaire. Douze implants ont e´te´ pose´s : cinq mandibulaires et sept maxillaires. Les re´sultats esthe´tiques et fonctionnels ont e´te´ bons, ame´liorant la qualite´ de vie. Discussion. La re´habilitation implanto-porte´e tre`s pre´coce est possible, voire souhaitable, lorsque les conditions sont re´unies chez ces patients. Elle re´tablit des fonctions oro-faciales qui favorisent le de´veloppement des bases squelettiques maxillo-faciales. Il existe des risques lie´s a` cette option the´rapeutique. Mais peut-on attendre jusqu’a` l’adolescence avec une prothe`se amovible non adapte´e, sous pre´texte d’une croissance non acheve´e ? ß 2013 Elsevier Masson SAS. Tous droits re´serve´s. Mots cle´s : Implant, Hypodontie, Dysplasie ectodermique
Introduction DOI of original article: http://dx.doi.org/10.1016/j.revsto.2013.01.009 * Corresponding author. E-mail addresses: [email protected], [email protected]
Ectodermal dysplasia includes a heterogeneous group of rare genetic diseases characterized by development abnormalities of ectodermal structures [1–7]. Anhidrotic ectodermal
2213-6533/$ - see front matter ß 2013 Elsevier Masson SAS. All rights reserved. http://dx.doi.org/10.1016/j.revsto.2013.03.008 Rev Stomatol Chir Maxillofac Chir Oral 2013;114:e5-e8
e5
C. Paulus, P. Martin
dysplasia is a rare disease combining hypodontia, hypotrichosis, and hypohidrosis. Currently, around environ 200 kinds of ectodermal dysplasia, different genetically and clinically have been identified. Anhidrotic ectodermal dysplasia is the most frequent of these ectodermal dysplasias (7/10,000 births). The patient’s quality of life is greatly impaired and this at a very early age. In most of cases, there are abnormalities on the number (anodontia, hypodontia) and shape (conical) of teeth. This often causes major oro-facial dysfunctions and social handicap. Usual dental prostheses are not adapted to a very young patient. Very early implant-borne prosthetic rehabilitation could be an alternative. We report the case of very early implant placement in a child presenting with hypodontia related to ectodermal dysplasia.
Rev Stomatol Chir Maxillofac Chir Oral 2013;114:e5-e8
Figure 2. Panoramic X-ray of two dystrophic germs, 21 and 11.
Observation A 23-month old child was brought to the pediatric stomatology and maxillo-facial surgery unit in January 2005, for ‘‘missing teeth’’. Patient history documented episodes of hyperthermia. An extra-oral examination allowed observing, thin and dry skin with periorbital and peri-buccal wrinkles, periorbital hyperpigmentation, thin and sparse hair fins, a bilateral labial commissural perleche, and a labial vertical height defect (fig. 1). The oral examination revealed three cone shaped canines (23, 33, and 43). The radiological assessment (panoramic X-ray, frontal and lateral teleradiography) allowed visualizing two dystrophic germs (21 and 11) (figs. 2 and 3). The diagnosis of ectodermal dysplasia was made on these findings. Dental rehabilitation was undertaken with removable dental devices. But the lack of stability required multiple adaptations
Figure 1. Thin and dry skin with periorbital and peri-buccal wrinkles, periorbital hyperpigmentation, thin and sparse hair fins, a bilateral labial commissural perleche, and a labial vertical height defect.
e6
Figure 3. Profile teleradiography revealing severe maxillo-mandibular bone resorption.
and several new prostheses (years 2006, 2007, and 2008). The onset of oral candidiasis contributed to the patient’s rejection of removable dental devices. In December 2008, when the child was 5 years and 9 months of age, it was decided to use maxillo-mandibular implantborne prosthetic rehabilitation, after obtaining the parents’ enlightened consent concerning the risks of this therapeutic strategy: multiple general anesthesia (GA) procedures and risk of graft or implant failure. The first GA took place in June 2009, when the child was 6 years old. Teeth 33 and 43 were extracted. Five implants, 3.4 mm in diameter and 13 mm long (Progress, TekkaW, Lyon, France), were placed in the site of teeth 34 to 44, after performing mucoperiosteal flaps. An autogenous bone graft was placed on the lingual and vestibular side of the symphysis. Tooth 13 and impacted 21 and 11 were extracted on the maxilla, after performing a crestal incision, posterior discharge incision, and mucoperiosteal debridement. Seven implants were placed:
Hypodontia due to ectodermal dysplasia
Figure 4. Final results after complete rehabilitation with significant esthetic improvement.
three with a diameter of 4 mm and 13 mm long (in tooth area 13, 14, and 16); three with a diameter of 3.4 mm and 13 mm long (in tooth area 23, 24, and 26); one with a diameter of 3.4 mm 10 mm long in tooth area 11.
Grafts associating autogenous bone and bank bone (OsteoPureW) were placed at the bilateral intra-sinus, vestibular, palatine, and peri-implant level. The wound was closed in two planes with Vicryl 3/0 (EthiconW) after sub-periosteal dissection and striation of the periosteum. Mandibular implants were exposed after 4 months, and transgingival screws were placed during a second GA. Multi-cone pillars were then inserted, as well as a trans-screwed beam. A Bra˚nemarkW type bridge was screwed. The removable superior prosthesis was fractured several times before placement of a fixed maxillary prosthesis, because of chewing on the fixed inferior prosthesis. The maxillary implants were exposed after 20 months, and trans-gingival screws were placed during a third GA. The print and the vertical dimension were evaluated during this third procedure. The final maxillary prosthetic rehabilitation was made with two trans-screwed ceramic and metallic hemi-bridges, freeing the palatine suture. The child immediately found the prostheses to be comfortable, esthetic, and especially efficient. Dental hygiene was significantly improved, and 2 months before the end of rehabilitation, the weight gain had reached 3 kgs, thanks to a rehabilitated chewing function. The phonation was normal; there was no lingual interposition, or infantile deglutition. Concerning the esthetic aspect, the labial ratios were normalized and the vertical deficiency resolved. The perleche and the peri-buccal wrinkles disappeared (fig. 4). The child became more calm and self-confident. His schooling returned to normal and he no longer had any relational problems.
Figure 5. Results after two postoperative months a (panoramic) and b (profile).
The regular radiological follow-up revealed the stability of fixtures and a normal growth of the maxilla (fig. 5). None implant was lost.
Discussion The implementation of intra-osseous implants is often postponed until the end of maxillary and mandibular growth. Some authors have proposed an earlier implantation [1,3,6]. We decide to implement an early implantation, despite our patient’s age (6 years), because of a severe hypodontia severe preventing the use of stable removable prostheses. This had for consequences a great physiological, esthetic, and social impact. The major risks were: short-, medium-, or long-term implant failure; and repeated GAs. But the benefits seemed significantly superior to the risks: early implant placement allowed soliciting maxillo-facial growth thanks to rehabilitated muscular activity;
e7
C. Paulus, P. Martin
social integration was available thanks to esthetic, functional, and psychological improvement. These benefits had been confirmed by other authors [4,7,8].
The maxilla of children with ectodermal dysplasia presents atrophy and bone resorption related to severe dental agenesis [2,9]; it is not totally functional and not adapted to a removable prosthesis. There are no studies on early maxillary implantation. We suppose that implants act as ‘‘external fixtures’’ and stimulate the facial growth by rehabilitating oro-facial functions. Given the very young age, a regular radiological follow-up must be implemented to assess the growth and outcome of implants and peri-implant tissues. Prostheses on early implants should be considered as temporary. This type of treatment should be validated by future studies and long-term results assessed. The issue raised in this case is: ‘‘Should a patient have to endure non-adapted prostheses during his childhood and teenage, because growth is not finished?’’ One of the most interesting perspectives for patients presenting with hypodontia or anodontia is using pluripotent stem cells to induce a regeneration of dental organs [10].
Rev Stomatol Chir Maxillofac Chir Oral 2013;114:e5-e8
References [1]
[2]
[3]
[4]
[5]
[6]
[7]
[8]
[9]
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
e8
[10]
Bjork A, Skieller V. Growth of the maxilla in three dimensions as revealed radiographically by the implant method. Br J Orthod 1977;4:53–64. Bonin B, Saffarzadeh A, Picard A, Levy P, Romieux G, Goga D. Traitement implantaire pre´coce d’un enfant atteint d’une dysplasie ectodermique anhydrotique. A` propos d’un cas. Rev Stomatol Chir Maxillofac 2001;102:313–8. Davarpanah M, Moon JW, Yang LR, Celletti R, Martinez H. Dental implants in the oral rehabilitation of a teenager with hypohidrotic ectodermal dysplasia: report of a case. Int J Oral Maxillofac Implants 1997;12:252–8. Fotso J, Hugentobler M, Kiliaridis S, Richter M. Dysplasie ectodermique anhydrotique. Re´habilitation. Rev Stomatol Chir Maxillofac 2009;110:50–4. Pinheiro M, Freire-Maia N. Ectodermal dysplasias: a clinical classification and a causal review. Am J Med Genet 1994;53: 153–62. Guckes AD, McCarthy 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. 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. Kramer FJ, Baethge C, Tschernitscheck H. Implants in children with ectodermal dysplasia: a case report and literature review. Clin Oral Implants Res 2007;18:140–6. Soubeyrand E, Nicolas J, Labbe´ D, Riscala S, Olive L, Compe`re JF. La dysplasie ectodermique anhydrotique : pre´sentation de quatre observations. Rev Stomatol Chir Maxillofac 2005;106: 328–33. Magloire H, Couble ML. L’implant dentaire biologique : mythe ou re´alite´ ? Rev Stomatol Chir Maxillofac 2011;112:240–8.