Interdisciplinary treatment with implant-supported prostheses for an adolescent with ectodermal dysplasia: A clinical report

Interdisciplinary treatment with implant-supported prostheses for an adolescent with ectodermal dysplasia: A clinical report

CLINICAL REPORT Interdisciplinary treatment with implant-supported prostheses for an adolescent with ectodermal dysplasia: A clinical report Yajing L...

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CLINICAL REPORT

Interdisciplinary treatment with implant-supported prostheses for an adolescent with ectodermal dysplasia: A clinical report Yajing Liu, MDSa and Chunbo Tang, DDS, PhDb Ectodermal dysplasia (ED) is a ABSTRACT congenital syndrome with at This clinical report describes interdisciplinary treatments for a 17-year-old girl with ectodermal least 2 ectodermally derived dysplasia. The treatment was initiated with orthodontic therapy. After the remaining primary 1,2 dysplasia structures. Oral teeth had been extracted, 6 implants were placed in the maxilla with bilateral sinus floor abnormalities include hypoelevations, and 6 implants were placed in the mandible. Immediate restorations were provided. dontia, anodontia, and conical Definitive restorations included screw-retained partial dental prostheses and ceramic crowns. (J Prosthet Dent 2019;-:---) teeth.3,4 Furthermore, severe hypodontia may contribute to atrophic alveolar bone, abnormal jaw relationship, and skeletal maturation, implant-supported restorations are loss of vertical dimension, making implant-supported reliable treatments.15-19 As the oral manifestations are 5-10 restorations challenging. Removable prostheses are complex and changing, treatment plans should be the conventional prosthodontic treatment for children personalized, and an interdisciplinary team approach is because of their continuing skeletal growth.11-14 After necessary.20,21

Figure 1. Before treatment. A, Intraoral photograph. B, Panoramic radiograph.

This work was supported by National Science Foundation of China grant #81470778, the Science and Technology Commission Program of Nanjing grant #201605011, and the Southeast University and Nanjing Medical University Cooperative Research Project grant #2242018K3DN03. a Postgraduate student, Graduate Prosthodontics, Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, PR China; and Postgraduate student, Department of Dental Implantology, Nanjing Medical University, Nanjing, PR China. b Professor, Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, PR China; and Professor, Department of Dental Implantology, Affiliated Hospital of Stomatology, Nanjing Medical University, Nanjing, PR China.

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Figure 2. A, Occlusal device, orthodontic bands, elastics, and brackets during orthodontic treatment. B, Increased occlusal vertical dimension (5 mm) obtained.

Figure 3. Conditions after primary tooth extraction. A, Intraoral view. B, Panoramic radiograph.

Figure 4. A, Implant placement in mandible. B, Interim fixed prosthesis delivered immediately.

CLINICAL REPORT A 17-year-old adolescent with ED presented in 2014 with the chief complaint of an unesthetic appearance. The intraoral and radiographic examinations revealed only

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6 permanent teeth: maxillary central incisors and the 4 first molars. Primary teeth were present, including the molars, the canines, and the maxillary lateral incisors. The restorative space was reduced (Fig. 1). The treatment plan involved orthodontic treatment to provide adequate Liu and Tang

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Figure 5. A, Implants placement with transcrestal sinus floor elevation in maxilla. B, Interim prosthesis relined and delivered.

Figure 6. A, Implant-level impression made in maxilla. B, Screw-retained zirconia ceramic partial dental prostheses and crowns fabricated for maxilla. C, Abutment-level impression made with transfer copings splinted in mandible. D, Screw-retained metal-ceramic dental prosthesis fabricated for mandible.

occlusal vertical dimension and minimize the midline diastema, extraction of the primary teeth, implant placement, implant-supported fixed dental prostheses and ceramic crowns for the definitive restorations, and psychological support. Orthodontic treatment was initiated in 2014. An occlusal device was fabricated to provide space for the

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eruption of the permanent first molars with orthodontic bands (American Orthodontics) and elastics. Orthodontic brackets (American Orthodontics) were attached to the maxillary central incisors to close the midline diastema. After 12 months, an increased occlusal vertical dimension of 5 mm and a decreased midline diastema were achieved (Fig. 2). Lateral cephalograms were used to verify

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Figure 7. Definitive protheses. A, Maxillary. B, Frontal. C, Mandibular. D, Panoramic radiograph.

the patient’s skeletal maturity with the cervical vertebral maturation (CMV) method.22,23 In 2015, the remaining primary teeth were extracted with minimal trauma. Removable partial dentures had been prefabricated and were delivered as immediate interim prostheses. After 2 months of healing, a cone beam computed tomography (CBCT) scan showed sufficient residual bone volume in the mandible but inadequate bone volume in the maxillary posterior regions (Fig. 3). In the mandible, the interim prosthesis was modified into a surgical template for implant placement.24 Under local anesthesia, 6 implants (Ankylos Plus; Dentsply Sirona) were placed and abutments connected. The interim fixed prosthesis was delivered immediately (Fig. 4). In the maxilla, transcrestal sinus floor elevation was performed in the right posterior regions. Meanwhile, maxillary sinus floor elevation was performed with the lateral window technique on the left side with xenografts (Bio-Oss; Geistlich Pharma AG) and resorbable collagen membrane (Bio-Gide; Geistlich Pharma AG). Six implants (Ankylos Plus; Dentsply Sirona) were inserted in the maxilla by a 2stage approach. The interim maxillary prosthesis was then relined and delivered immediately (Fig. 5). In 2016, osseointegration was confirmed from radiographs. The maxillary implants were uncovered, and the

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healing abutments attached. After 2 weeks, the maxillary central incisors were prepared for ceramic crowns and definitive impressions made with polyether material (Impregum; 3M ESPE). The ceramic crowns (Ceramill Zirconia; Amann Girrbach AG) and zirconia ceramic partial dental prostheses were cemented (Panavia F2.0; Kuraray) on the abutments (Ankylos; Dentsply Sirona) extraorally with screw-access holes on the occlusal surfaces. A cobalt-chromium framework was fabricated for the mandible, passive fit was verified intraorally, and a screw-retained metal-ceramic dental prosthesis was fabricated (Fig. 6).25-27 At delivery, adjustments were made to reach balanced occlusion and reduce the occlusal contact on the maxillary cantilevers. The definitive restoration was completed when the patient was 19 years old (Fig. 7). During the treatment, the patient’s emotions were closely monitored.28-30 Timely encouragements and psychological counseling were also provided. During a 2year follow-up period, professional recall visits were conducted at 6-month intervals.31,32 Good function and esthetics were observed. Radiological examination revealed that all the implants had osseointegrated with no measurable marginal bone loss. However, gingival inflammation was present in the maxillary incisor region, Liu and Tang

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Figure 8. Two years after treatment. A, Intraoral view. B, Panoramic radiograph.

which was addressed with professional oral hygiene maintenance (Fig. 8).33,34 DISCUSSION Information about the clinical outcomes of implants in patients with ED is sparse,7-9,17 although relatively low failure similar to those without ED has been reported.15 However, others have reported a slightly lower survival rate.8,9 Bone deficiencies and diminished vascularity may contribute to the failure.10 More information is needed regarding long-term outcomes of implants placed in patients with ED. Removable complete dentures have poor retention in patients with ED,7 while implant-supported prostheses have advantages in both functional and esthetical aspects,16 although biological and technical complications frequently occur.33 These complications may not affect the success of the definitive restorations, but they do increase maintenance costs.34 Implant placement should be delayed until skeletal maturation is complete or nearly complete.11-14 In young patients, changes in anatomic morphology may require modification or replacement of prostheses; therefore, screw-retained prostheses with greater retrievability25-27 are preferred.26 The psychosocial impact of ED is often overlooked, with patients often having emotional problems.28,29 Saltnes et al30 reported that the levels and prevalence of anxiety were higher in patients with ED, and almost half of them had anxiety scores above the level requiring additional clinical examination. Psychological treatment is an integral part of the overall treatment plan, and psychological testing, counseling, or appropriate referral should be provided when necessary. SUMMARY The interdisciplinary treatment for the adolescent with ectodermal dysplasia included orthodontic treatment, Liu and Tang

bone grafting, implant placement, prosthetic treatment, periodontal treatment, and psychological support. No significant complications occurred during the 2-year follow-up, demonstrating successful restorative care with an interdisciplinary approach. REFERENCES 1. Salinas CF, Irvine AD, Itin PH, Di Giovanna JJ, Schneider H, Clarke AJ, et al. Second International Conference on a classification of ectodermal dysplasias: development of a multiaxis model. Am J Med Genet A 2014;164:2482-9. 2. Pagnan NA, Visinoni AF. Update on ectodermal dysplasias clinical classification. Am J Med Genet A 2014;164:2415-23. 3. Khalaf K, Miskelly J, Voge E, Macfarlane TV. Prevalence of hypodontia and associated factors: a systematic review and meta-analysis. J Orthod 2014;41: 299-316. 4. Dhamo B, Kuijpers MAR, Balk-Leurs I, Boxum C, Wolvius EB, Ongkosuwito EM. Disturbances of dental development distinguish patients with oligodontia-ectodermal dysplasia from isolated oligodontia. Orthod Craniofac Res 2018;21:48-56. 5. Rajan G, Mariappan S, Ramasubramanian H, Somasundaram S, Natarajarathinam G. Restoration of atrophic edentulous maxilla of a patient with ectodermal dysplasia using quadruple zygomatic implants: a case report. J Maxillofac Oral Surg 2015;14:848-52. 6. Schnabl D, Grunert I, Schmuth M, Kapferer-Seebacher I. Prosthetic rehabilitation of patients with hypohidrotic ectodermal dysplasia: a systematic review. J Oral Rehabil 2018;45:555-70. 7. Wang Y, He J, Decker AM, Hu JC, Zou D. Clinical outcomes of implant therapy in ectodermal dysplasia patients: a systematic review. Int J Oral Maxillofac Surg 2016;45:1035-43. 8. Yap AK, Klineberg I. Dental implants in patients with ectodermal dysplasia and tooth agenesis: a critical review of the literature. Int J Prosthodont 2009;22:268-76. 9. Huang PY, Driscoll CF. From childhood to adulthood: oral rehabilitation of a patient with ectodermal dysplasia. J Prosthet Dent 2014;112:439-43. 10. Bayat M, Khobyari MM, Dalband M, Momen-Heravi F. Full mouth implant rehabilitation of a patient with ectodermal dysplasia after orthognathic surgery, sinus and ridge augmentation: a clinical report. J Adv Prosthodont 2011;3:96-100. 11. Alsayed HD, Alqahtani NM, Alzayer YM, Morton D, Levon JA, Baba NZ. Prosthodontic rehabilitation with monolithic, multichromatic, CAD-CAM complete overdentures in an adolescent patient with ectodermal dysplasia: A clinical report. J Prosthet Dent 2018;119:873-8. 12. Machado M, Wallace C, Austin B, Deshpande S, Lai A, Whittle T, et al. Rehabilitation of ectodermal dysplasia patients presenting with hypodontia: outcomes of implant rehabilitation part 1. J Prosthodont Res 2018;62:473-8. 13. Knobloch LA, Larsen PE, Saponaro PC, L’Homme-Langlois E. Early implant placement for a patient with ectodermal dysplasia: Thirteen years of clinical care. J Prosthet Dent 2018;119:702-9. 14. Kramer FJ, Baethge C, Tschernitschek H. Implants in children with ectodermal dysplasia: a case report and literature review. Clin Oral Implants Res 2007;18:140-6. 15. Chrcanovic BR. Dental implants in patients with ectodermal dysplasia: a systematic review. J Craniomaxillofac Surg 2018;46:1211-7.

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16. Oh SH, Kim Y, Park JY, Jung YJ, Kim SK, Park SY. Comparison of fixed implant-supported prostheses, removable implant-supported prostheses, and complete dentures: patient satisfaction and oral health-related quality of life. Clin Oral Implants Res 2016;27:e31-7. 17. Moshaverinia A, Torbati A, Kar K, Aalam AA, Takanashi K, Chee WW. Full mouth rehabilitation of a young patient with partial expressions of ectodermal dysplasia: a clinical report. J Prosthet Dent 2014;112: 449-54. 18. Shi JY, Zhang XM, Qiao SC, Qian SJ, Mo JJ, Lai HC. Hardware complications and failure of three-unit zirconia-based and porcelain-fused-metal implantsupported fixed dental prostheses: a retrospective cohort study with up to 8 years. Clin Oral Implants Res 2017;28:571-5. 19. Papaspyridakos P, Barizan Bordin T, Kim YJ, DeFuria C, Pagni SE, Chochlidakis K, et al. Implant survival rates and biologic complications with implant-supported fixed complete dental prostheses: A retrospective study with up to 12-year follow-up. Clin Oral Implants Res 2018;29: 881-93. 20. Van Sickels JE, Raybould TP, Hicks EP. Interdisciplinary management of patients with ectodermal dysplasia. J Oral Implantol 2010;36:239-45. 21. Klineberg I, Cameron A, Hobkirk J, Bergendal B, Maniere MC, King N, et al. Rehabilitation of children with ectodermal dysplasia. Part 2: an international consensus meeting. Int J Oral Maxillofac Implants 2013;28:1101-9. 22. Baccetti T, Franchi L, McNamara JA Jr. An improved version of the cervical vertebral maturation (CVM) method for the assessment of mandibular growth. Angle Orthod 2002;72:316-23. 23. McNamara JA Jr, Franchi L. The cervical vertebral maturation method: a user’s guide. Angle Orthod 2018;88:133-43. 24. Kutkut A, Abu-Eid R, Sharab L, Abadi B, Van Sickels J. Full mouth implantsupported rehabilitation of a patient with ectodermal dysplasia: clinical report and literature review. J Int Acad Periodontol 2015;17:34-41. 25. Tallarico M, Caneva M, Baldini N, Gatti F, Duvina M, Billi M, et al. Patientcentered rehabilitation of single, partial, and complete edentulism with cemented- or screw-retained fixed dental prosthesis: The First Osstem Advanced Dental Implant Research and Education Center Consensus Conference 2017. Eur J Dent 2018;12:617-26. 26. Wittneben JG, Joda T, Weber HP, Brägger U. Screw retained vs. cement retained implant-supported fixed dental prosthesis. Periodontol 2000 2017;73:141-51. 27. Thoma DS, Wolleb K, Bienz SP, Wiedemeier D, Hämmerle CHF, Sailer I. Early histological, microbiological, radiological, and clinical response to cemented and screw-retained all-ceramic single crowns. Clin Oral Implants Res 2018;29:996-1006.

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28. Kohli R, Levy S, Kummet CM, Dawson DV, Stanford CM. Comparison of perceptions of oral health-related quality of life in adolescents affected with ectodermal dysplasias relative to caregivers. Spec Care Dentist 2011;31:88-94. 29. Saltnes SS, Jensen JL, Sæves R, Nordgarden H, Geirdal AØ. Experiences of daily life and oral rehabilitation in oligodontia - a qualitative study. Acta Odontol Scand 2019;77:197-204. 30. Saltnes SS, Jensen JL, Sæves R, Nordgarden H, Geirdal AØ. Associations between ectodermal dysplasia, psychological distress and quality of life in a group of adults with oligodontia. Acta Odontol Scand 2017;75:564-72. 31. Monje A, Aranda L, Diaz KT, Alarcón MA, Bagramian RA, Wang HL, et al. Impact of maintenance therapy for the prevention of peri-implant diseases: a systematic review and meta-analysis. J Dent Res 2016;95:372-9. 32. Bidra AS, Daubert DM, Garcia LT, Kosinski TF, Nenn CA, Olsen JA. Clinical practice guidelines for recall and maintenance of patients with tooth-borne and implant-borne dental restorations. J Prosthodont 2016;25(Suppl 1): S32-40. 33. Wittneben JG, Buser D, Salvi GE, Bürgin W, Hicklin S, Brägger U. Complication and failure rates with implant-supported fixed dental prostheses and single crowns: a 10-year retrospective study. Clin Implant Dent Relat Res 2014;16:356-64. 34. Le M, Papia E, Larsson C. The clinical success of tooth- and implantsupported zirconia-based fixed dental prostheses. A systematic review. J Oral Rehabil 2015;42:467-80. Corresponding author: Dr Chunbo Tang Department of Dental Implantology Affiliated Hospital of Stomatology Nanjing Medical University 136 Hanzhong Rd Nanjing, Jiangsu 210029 PR CHINA Email: [email protected] Acknowledgments The authors thank American Orthodontics, Geistlich Pharma AG, Tokuyama Dental Corp, and 3M ESPE for their materials, especially Dentsply Sirona for their implant components. Copyright © 2019 by the Editorial Council for The Journal of Prosthetic Dentistry. https://doi.org/10.1016/j.prosdent.2019.07.006

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