Dental Implant Therapy for a Child with X-linked Hypohidrotic Ectodermal Dysplasia— Three Decades of Managed Care
Abstract
Purpose The aim was to report on oral rehabilitation of a boy with X-linked hypohidrotic ectodermal dysplasia (XLHED) and anodontia of the mandible between ages 3 and 33 years where treatment involved dental implants and oral care management by a multidisciplinary team of specialists. Materials and methods This case history report describes the clinical management of a boy born in 1979 with XLHED and anodontia of the mandible. Two implants were successfully placed in the anterior region of the mandible in 1985. Genetic analysis later verified the diagnosis by confirming a mutation in the EDA gene. The case description was based on review of the patient records and assessment of orofacial function. Results The patient had satisfactory orofacial appearance and function throughout his youth. He is still caries-free at age 33 and has experienced only minor oral complications. Dental management began at age 3, when he received a maxillary removable dental prosthesis. At age 7, he received a mandibular implant-supported overdenture. After two more implants in the mandible and orthodontic treatment in the maxilla, his oral rehabilitation was completed at age 22 with maxillary tooth-supported and mandibular implant-supported fixed dental prostheses. Regular follow-ups provided supervision of oral hygiene, caries prevention, and prosthetic maintenance. Conclusion This long-term follow-up of a child with XLHED and anodontia in the mandible supports the use of dental implants, with cons…
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In children aged six to fourteen years, mandibular implant survival times of one up to 26 years were reported.
“In children aged six to fourteen years, mandibular implant survival times of one up to 26 years were reported.26,31,65,66,73,89”
From Prosthetic rehabilitation of patients with hypohidrotic ectodermal dysplasia: A systematic review (2018) · cites this paper as [31]
The case report by Bergendal 1991 with a follow-up of 9 years was described again by Bergendal 2015 with a follow-up of 29 years.
“the case report by Bergendal 1991 84 with a follow-up of 9 years was described again by Bergendal 2015 31 with a follow-up of 29 years.”
From Prosthetic rehabilitation of patients with hypohidrotic ectodermal dysplasia: A systematic review (2018) · cites this paper as [31]
One individual received an implant-supported overdenture with ball attachments in the mandibula at age 6 years.
“one individual received an implant-supported overdenture with ball attachments in the mandibula at age 6 years.31”
From Prosthetic rehabilitation of patients with hypohidrotic ectodermal dysplasia: A systematic review (2018) · cites this paper as [31]
Renewal of partial dentures has to be expected after one to 6 years.
“and renewal of partial dentures after one to 6 years.24,31,[85][86][87][88]”
From Prosthetic rehabilitation of patients with hypohidrotic ectodermal dysplasia: A systematic review (2018) · cites this paper as [31]
The spectrum of long-term follow-up in included reports ranges from reporting on the achievement of dental treatment without further monitoring to less frequent reports with more than a decade of monitoring.
“The spectrum ranges from reporting on the achievement of dental treatment without further monitoring to less frequent reports with more than a decade of monitoring (e.g., Bergendal et al. 2015; Fernandes et al. 2016; James and Roudsari 2019).”
From Dental Management of Genetic Dental Disorders: A Critical Review (2025) · cites this paper as Bergendal et al. 2015
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Clinical outcomes of implant therapy in ectodermal dysplasia patients: a systematic review
Wang, Y., He, J., Decker, A. M., Hu, J. C. ‐., & Zou, D. (2016) · International Journal of Oral and Maxillofacial Surgery · doi:10.1016/j.ijom.2016.03.011 · Cited by 72 · see citing works
Prosthetic rehabilitation of patients with hypohidrotic ectodermal dysplasia: A systematic review
Schnabl, D., Grunert, I., Schmuth, M., & Kapferer‐Seebacher, I. (2018) · Journal of Oral Rehabilitation · doi:10.1111/joor.12638 · Cited by 63 · see citing works
“Eight case reports (20, 25, 31, 83-88) and two case series (24, 89) described the long-term care of 17 HED-individuals (14 males, 3 females) with several prosthetic follow-up treatments (Table 5); the case report by Bergendal 1991 (84) with a follow-up of nine years was described again by Bergendal 2015 (31) with a fo…”
BackgroundInfluentialDental implants in growing patients: A systematic review and meta-analysis
Elagib, M. F. A., Alqaysi, M. A. H., Almushayt, M. O. S., Nagate, R. R., Gokhale, S. T., & Chaturvedi, S. (2023) · Technology and Health Care · doi:10.3233/thc-220581 · Cited by 35 · see citing works
Dental implant considerations in patients with systemic diseases: An updated comprehensive review
Mosaddad, S. A., Talebi, S., Keyhan, S. O., Fallahi, H. R., Darvishi, M., Aghili, S. S., Tavahodi, N., Namanloo, R. A., Heboyan, A. G., & Fathi, A. (2024) · Journal of Oral Rehabilitation · doi:10.1111/joor.13683 · Cited by 23 · see citing works
Cerezo-Cayuelas, M., Pérez-Silva, A., Serna-Muñoz, C., Vicente, A., Martínez-Beneyto, Y., Cabello-Malagón, I., & Ortiz-Ruíz, A. (2022) · Orphanet Journal of Rare Diseases · doi:10.1186/s13023-022-02533-0 · Cited by 18 · see citing works
Machado, M., Wallace, C., Austin, B., Deshpande, S., Lai, A., Whittle, T., & Klineberg, I. J. (2018) · Journal of Prosthodontic Research · doi:10.1016/j.jpor.2018.07.001 · Cited by 18 · see citing works
Implant Supported Oral Rehabilitation in a 5 Year Old Ectodermal Dysplasia Patient
Rajan, G., Mariappan, S., Balachandran, A., Krishnamoorthy, S., & Muthukuru, M. (2019) · Clinical Dentistry · doi:10.33882/clinicaldent.13.25192 · Cited by 15 · see citing works
Oligodontia in the Clinical Spectrum of Syndromes: A Systematic Review
Castilho, N. L., Resende, K. K. M., dos Santos, J. A., Machado, R. A., Coletta, R. D., Guerra, E. N. S., Acevedo, A. C., & Martelli‐Júnior, H. (2023) · Dentistry Journal · doi:10.3390/dj11120279 · Cited by 15 · see citing works
Casaña-Ruiz, M. D., Catalá‐Pizarro, M., Borrás-Aviñó, C., Sanchis, M. E., & Bellot‐Arcís, C. (2023) · Journal of Clinical and Experimental Dentistry · doi:10.4317/jced.60168 · Cited by 10 · see citing works
Dental Management of Genetic Dental Disorders: A Critical Review
Dujic, H., Bücher, K., Schüler, I. M., Schmidt, P., Hertel, S., Timpel, J., Jablonski-Momeni, A., Schilke, R., Kapferer-Seebacher, I., Zschocke, J., et al. (2025) · Journal of Dental Research · doi:10.1177/00220345241305330 · Cited by 8 · see citing works
“The spectrum ranges from reporting on the achievement of dental treatment without further monitoring to less frequent reports with more than a decade of monitoring (e.g., Bergendal et al. 2015; Fernandes et al. 2016; James and Roudsari 2019).”
Barve, K. D., Padawe, D. S., & Takate, V. S. (2025) · International Journal of Clinical Pediatric Dentistry · doi:10.5005/jp-journals-10005-3069 · Cited by 7 · see citing works
Sinada, N. G., & Wang, C. I. (2021) · Journal of Prosthetic Dentistry · doi:10.1016/j.prosdent.2021.07.022 · Cited by 4 · see citing works
Other Conditions Affecting Dental Implant Treatment
Duan, X., Doi, K., Yuan, Q., & Zhang, S. (2019) · Springer eBooks · doi:10.1007/978-3-030-28557-9_10 · Cited by 3 · see citing works
Arte, S., Awadh, W., Nieminen, P., & Rice, D. P. C. (2017) · Springer eBooks · doi:10.1007/978-3-319-54637-7_5 · Cited by 3 · see citing works
Interdisciplinary management of Type 3 oligodontia: A retrospective case series
Liddelow, G. J., Singer, S. L., Allan, B. P., & Henry, P. J. (2024) · Journal of Prosthetic Dentistry · doi:10.1016/j.prosdent.2024.05.028 · Cited by 1 · see citing works
Alsharbaty, M. H. M., & Bahrami, M. (2019) · Dental Hypotheses · doi:10.4103/denthyp.denthyp_9_19 · Cited by 1 · see citing works
Tosun, T., & Koray, M. (2019) · IntechOpen eBooks · doi:10.5772/intechopen.81202
Gjurin, S. Z., Hren, N. I., Tomaževič, T., & Oblak, Č. (2020) · Slovenian Medical Journal · doi:10.6016/zdravvestn.3004
Chochlidakis, K., Lampraki, E., Carranza, M. G., Ercoli, C., Nikellis, T., & Tsigarida, A. (2021) · Journal of the California Dental Association · doi:10.1080/19424396.2021.12222720
Dental Implants and Systemic Diseases
Mosaddad, S. A., Keyhan, S. O., Fallahi, H. R., Darvishi, M., Aghili, S. S., & Namanloo, R. A. (2024) · Springer eBooks · doi:10.1007/978-3-031-36962-9_16-1
Implant-supported fixed prosthesis for orthognathic surgery in ectodermal dysplasia: a case report
Shin, Y.-A., Moon, J.-E., Kang, S.-H., Park, C.-I., Bae, Y., Oh, M.-S., Jeon, W., Kang, N., & Baek, M.-J. (2025) · The Journal of Korean Academy of Prosthodontics · doi:10.4047/jkap.2025.63.1.20
Ponomarev, A. E., Butsan, S. B., Iigitaliev, S. N., Lafishev, A. I., Bulat, S. G., & Seleznev, V. A. (2025) · Stomatology · doi:10.17116/stomat202510405164
Marcella, S., Sirica, R., Petrillo, N., Ianniello, M., Mori, A., Castiello, R., Capone, S. F., Evangelista, E., Suero, T., Ruggiero, R., et al. (2025) · Genes · doi:10.3390/genes16121484
Cerezo-Cayuelas, M., Silva, A. P., Muñoz, C. S., Vicente, A., Beneyto, Y. M., Cabello, I., & Ortiz-Ruiz, A. J. (2022) · Figshare · doi:10.6084/m9.figshare.21351265
Nakaoka, K., Yamada, S., Sato, K., Ishizuka, T., Hojo, H., & Hamada, Y. (2025) · Journal of Oral and Maxillofacial Surgery Medicine and Pathology · doi:10.1016/j.ajoms.2025.09.006
Saini, A., Gupta, R., Gill, S., Mishra, A., Choudhary, P., & Kaushik, A. (2026) · Cureus · doi:10.7759/cureus.118409
Αντωνιάδης, Κ., Kyriakou, N., & Kyriakidou, E. (2026) · Journal of Cranio-Maxillofacial Surgery · doi:10.1016/j.jcms.2026.109931
- Authors
- Bergendal, B., Bjerklin, K., Bergendal, T., & Koch, G.
- Venue
- The International Journal of Prosthodontics
- Publisher
- Quintessence Publishing
- Published
- 2015-07
- Volume
- 28
- Issue
- 4
- Pages
- 348-356
- ISSN
- 0893-2174 (ISSN-L)
- OpenAlex
- W2405185624
- Cited by
- 28
- References
- 0
- Citations by year
- 2026: 2 · 2025: 6 · 2024: 3 · 2023: 2 · 2022: 3 · 2021: 2 · 2020: 1 · 2019: 5 · 2018: 2 · 2017: 1 · 2016: 1
- FWCI
- 0.58
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- The International Journal of Prosthodontics (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › dental development and anomalies
- Health Sciences › Dentistry › Oral Surgery › Dental Implant Techniques and Outcomes
- Health Sciences › Health Professions › Emergency Medical Services › Dental Trauma and Treatments
- Keywords
- X-linked hypohidrotic ectodermal dysplasia, dental implants, anodontia, oral rehabilitation, implant-supported overdentures, removable dentures, dental caries prevention, oral hygiene, long-term follow-up, managed care, multidisciplinary dental treatment, orofacial function
- MeSH terms
- Child, Preschool, Humans, Infant, Managed Care Programs, Dental Implants, Ectodermal Dysplasia 1, Anhidrotic
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