{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T01:41:56Z","timestamp":1760146916622,"version":"build-2065373602"},"reference-count":39,"publisher":"MDPI AG","issue":"1","license":[{"start":{"date-parts":[[2024,12,26]],"date-time":"2024-12-26T00:00:00Z","timestamp":1735171200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>Background: The orthodontic management of pediatric patients with rare diseases, such as Ectodermal Dysplasia (ED) and Osteogenesis Imperfecta (OI), requires complex protocols due to dental anomalies in both the number and structure of teeth. These conditions necessitate a departure from traditional orthodontic approaches, as skeletal anchoring is often required because of these anomalies. Case Presentation: A patient with ED, characterized by hypodontia and malformed teeth, presented with insufficient natural teeth for anchorage. This challenge was addressed using a Maxillary Skeletal Expander (MSE) with miniscrews. Cone-beam computed tomography (CBCT) and cephalometric radiographs were used to assess bone density, which guided the creation of a customized hybrid device. A second patient with OI, a condition causing fragile bones, had malformed teeth and a high risk of fractures. Skeletal anchoring with MSE and miniscrews was chosen to avoid damaging brittle bones. The fragile nature of the patient\u2019s bones required careful planning and close monitoring throughout the treatment process. Both patients were treated at the UOC of Pediatric Dentistry, Sapienza University of Rome, using MSE with miniscrews. Pre- and post-treatment imaging (CBCT and cephalometric radiographs) were used to evaluate bone quality and monitor progress. Skeletal anchoring successfully addressed the unique challenges in both cases, achieving outcomes comparable to those in unaffected patients. Discsussions: despite limited bone volume, MSE successfully achieved maxillary arch expansion and improved occlusion. Post-treatment radiographs showed successful maxillary expansion and alignment without complications. Conclusions: This case series highlighted the effectiveness of MSE with miniscrews in treating patients with rare diseases. It advances orthodontic management by offering reliable solutions for complex cases involving dental anomalies and compromised bone structures.<\/jats:p>","DOI":"10.3390\/jcm14010055","type":"journal-article","created":{"date-parts":[[2024,12,26]],"date-time":"2024-12-26T04:31:16Z","timestamp":1735187476000},"page":"55","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Orthodontic Management in Pediatric Patients with Rare Diseases: Case Reports"],"prefix":"10.3390","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2502-6077","authenticated-orcid":false,"given":"Valeria","family":"Luzzi","sequence":"first","affiliation":[{"name":"Department of Oral and Maxillo-Facial Sciences, Sapienza University of Rome, U.O.C. Pediatric Dentistry Unit, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2041-2075","authenticated-orcid":false,"given":"Miriam","family":"Fioravanti","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillo-Facial Sciences, Sapienza University of Rome, U.O.C. Pediatric Dentistry Unit, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lilia","family":"Mitrano","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillo-Facial Sciences, Sapienza University of Rome, U.O.C. Pediatric Dentistry Unit, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Beatrice","family":"Marasca","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillo-Facial Sciences, Sapienza University of Rome, U.O.C. Pediatric Dentistry Unit, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3635-281X","authenticated-orcid":false,"given":"Matteo","family":"Saccucci","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillo-Facial Sciences, Sapienza University of Rome, U.O.C. Pediatric Dentistry Unit, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mauro","family":"Celli","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Sapienza University of Rome, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8657-8118","authenticated-orcid":false,"given":"Luca","family":"Celli","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Sapienza University of Rome, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8545-9889","authenticated-orcid":false,"given":"Iole","family":"Vozza","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillo-Facial Sciences, Sapienza University of Rome, U.O.C. Pediatric Dentistry Unit, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gaetano","family":"Ierardo","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillo-Facial Sciences, Sapienza University of Rome, U.O.C. Pediatric Dentistry Unit, 00161 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2024,12,26]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"965","DOI":"10.1111\/ijd.14048","article-title":"Hypohidrotic ectodermal dysplasia: Clinical and molecular review","volume":"57","year":"2018","journal-title":"Int. J. Dermatol."},{"key":"ref_2","first-page":"207","article-title":"Use of mini dental implants in ectodermal dysplasia children: Follow-up of three cases","volume":"15","author":"Sfeir","year":"2014","journal-title":"Eur. J. Paediatr. Dent."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"689","DOI":"10.1016\/j.jdent.2013.06.004","article-title":"Impact of moderate and severe hypodontia and amelogenesis imperfecta on quality of life and self-esteem of adult patients","volume":"41","author":"Hashem","year":"2013","journal-title":"J. Dent."},{"key":"ref_4","first-page":"140","article-title":"Ectodermal dysplasia: Important role of complex dental care in its interdisciplinary management","volume":"23","author":"Kratochvilova","year":"2022","journal-title":"Eur. J. Paediatr. Dent."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"1657","DOI":"10.1016\/S0140-6736(15)00728-X","article-title":"Osteogenesis imperfecta","volume":"387","author":"Forlino","year":"2016","journal-title":"Lancet"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"434","DOI":"10.1007\/s00774-011-0331-3","article-title":"Children and adolescents treated with neridronate for osteogenesis imperfecta show no evidence of any osteonecrosis of the jaw","volume":"30","author":"Maines","year":"2012","journal-title":"J. Bone Miner. Metab."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"314","DOI":"10.1111\/odi.13715","article-title":"Malocclusion in individuals with osteogenesis imperfecta: A systematic review and meta-analysis","volume":"28","author":"Prado","year":"2022","journal-title":"Oral Dis."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"19","DOI":"10.1002\/cre2.53","article-title":"Occlusal features and need for orthodontic treatment in persons with osteogenesis imperfecta","volume":"3","author":"Nguyen","year":"2017","journal-title":"Clin. Exp. Dent. Res."},{"key":"ref_9","doi-asserted-by":"crossref","unstructured":"Shroff, B., and Lindauer, S.J. (2010, December 27). Temporary Anchorage Devices: Biomechanical Opportunities and Challenges. In Current Therapy in Orthodontics; CHAPTER 22\u2014Temporary Anchorage Devices: Biomechanical Opportunities and Challenges; pp. 278\u2013290. Available online: https:\/\/typeset.io\/papers\/chapter-22-temporary-anchorage-devices-biomechanical-2e1aoi2si5.","DOI":"10.1016\/B978-0-323-05460-7.00022-3"},{"key":"ref_10","first-page":"539","article-title":"Mini-implants: Where are we?","volume":"39","author":"Melsen","year":"2005","journal-title":"J. Clin. Orthod."},{"key":"ref_11","doi-asserted-by":"crossref","unstructured":"Carvalho Ferreira, F.P., Barbosa Lima, A.P., de Paula, E.C., Ferreira Conti, A.C., Valarelli, D.P., and de Almeida-Pedrin, R.R. (2016). Orthodontic Protocol Using Mini-Implant for Class II Treatment in Patient with Special Needs. Case Rep. Dent., 2016.","DOI":"10.1155\/2016\/1057263"},{"key":"ref_12","doi-asserted-by":"crossref","unstructured":"Hartono, N., Soegiharto, B.M., and Widayati, R. (2018). The difference of stress distribution of maxillary expansion using rapid maxillary expander (RME) and maxillary skeletal Expander (MSE)\u2014A finite element analysis. Prog. Orthod., 19.","DOI":"10.1186\/s40510-018-0229-x"},{"key":"ref_13","unstructured":"Annarumma, F., Aghazada, H., and Pandolfi, S. (2024, November 13). L\u2019espansione Scheletrica Mascellare in Epoca Post-Puberale Attraverso L\u2019utilizzo delle Miniviti Palatali. Ortho Specialized. Available online: https:\/\/www.ortospecialized.it\/wp-content\/uploads\/2018\/03\/Espansione-schelettrica4.pdf."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1016\/j.ajodo.2007.10.045","article-title":"Buccal cortical bone thickness fr mini-implant placement","volume":"136","author":"Baumgaertel","year":"2009","journal-title":"Am. J. Orthod. Dentofac. Orthop."},{"key":"ref_15","first-page":"212","article-title":"Clinical applications of the Mini-Screw-Anchorage-System (M.A.S.) in the maxillary alveolar bone","volume":"5","author":"Carano","year":"2004","journal-title":"Prog. Orthod."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"230","DOI":"10.2319\/040309-192.1","article-title":"Changes in occlusal relationships in mixed dentition patients treated with rapid maxillary expansion. A prospective clinical study","volume":"80","author":"McNamara","year":"2010","journal-title":"Angle Orthod."},{"key":"ref_17","unstructured":"Derton, N., Gracco, A., Greco, A.M., and Luzi, C. (2015). Le Miniviti Ortodontiche\u2014Guida All\u2019utilizzo Clinico\u2014Edizioni Martina, Elsevier Science. Available online: https:\/\/www.infona.pl\/resource\/bwmeta1.element.elsevier-45b36db9-b8a0-3788-9db8-c77557123d7e."},{"key":"ref_18","unstructured":"Moyers Robert, E. (1991). Manuale di Ortodonzia, Scienza Tecnica Dentistica. [4th ed.]. Available online: https:\/\/www.medicalinformation.it\/manuale-di-ortodonzia-moyers.html."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1053\/j.sodo.2004.11.002","article-title":"Temporary devices in orthodontic: A paradigm shift","volume":"11","author":"Cope","year":"2005","journal-title":"Semin. Orthod."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"443","DOI":"10.1007\/s00056-007-0725-y","article-title":"Skeletal anchorage in orthodontics\u2014Basics and clinical application","volume":"68","author":"Wehrbein","year":"2007","journal-title":"J. Orofac. Orthop."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"9","DOI":"10.1093\/ejo\/cjy015","article-title":"Success of palatal implants or miniscrews placed median or paramedian for the reinforcement of anchora during orthodontic treatment: A systematic review","volume":"41","author":"Kakali","year":"2019","journal-title":"Eur. J. Orthod."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"595","DOI":"10.1034\/j.1600-0501.2000.011006595.x","article-title":"Alternative to the median region of the palate for placement of an orthodontic implant","volume":"11","author":"Bernhart","year":"2000","journal-title":"Clin. Oral Implant. Res."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"e6","DOI":"10.1016\/j.tripleo.2006.11.022","article-title":"The use of miniscrews implants for temporary skeletal anchorage in orthodontics: A comprehensive review","volume":"103","author":"Papadopoulos","year":"2007","journal-title":"Oral Surg. Oral Med. Oral Pathol. Oral Radiol."},{"key":"ref_24","first-page":"e70","article-title":"Temporary anchorage deviced (TADs) in orthodontics: Review of the factors that influence the clinical success rate of the mini-implants","volume":"167","author":"Leo","year":"2016","journal-title":"Clin. Ter."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"108","DOI":"10.1186\/s13023-024-03096-y","article-title":"The impact of osteogenesis imperfecta severity on oral health-related quality of life in Spain: A cross-sectional study","volume":"19","author":"Elfituri","year":"2024","journal-title":"Orphanet. J. Rare Dis."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"388","DOI":"10.1093\/ejo\/cjq090","article-title":"Factors influencing the stability of miniscrews. A retrospective study on 300 miniscrews","volume":"33","author":"Manni","year":"2011","journal-title":"Eur. J. Orthod."},{"key":"ref_27","first-page":"e168","article-title":"Using of modified rapid palate expander with miniscrews in a patient affected by ectodermic dysplasia","volume":"170","author":"Ierardo","year":"2019","journal-title":"Clin Ter."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1016\/j.ajodo.2008.08.032","article-title":"Relationship between vertical skeletal pattern and success rate of orthodontic mini-implants","volume":"138","author":"Moon","year":"2010","journal-title":"Am. J. Orthod. Dentofac. Orthop."},{"key":"ref_29","first-page":"348","article-title":"miniscrew-supported intrusion auxiliary for open-bite treatment with Invisalign","volume":"48","author":"Giancotti","year":"2014","journal-title":"J. Clin. Orthod."},{"key":"ref_30","first-page":"75","article-title":"Skeletal anchorage in orthodontics--a review of various systems in animal and human studies","volume":"23","author":"Janssen","year":"2008","journal-title":"Int. J. Oral Maxillofac. Implant."},{"key":"ref_31","first-page":"627","article-title":"Palatal-implant anchorage in an adult class II patient","volume":"45","author":"Giancotti","year":"2011","journal-title":"J. Clin. Orthod."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"174","DOI":"10.1111\/j.1708-8208.2008.00072.x","article-title":"Clinical outcome of mini-screws used as orthodontic anchorage","volume":"10","author":"Justens","year":"2008","journal-title":"Clin. Implant. Dent. Relat. Res."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"194","DOI":"10.1016\/j.ajodo.2008.03.031","article-title":"Survival analysis of orthodontic mini-implants","volume":"137","author":"Lee","year":"2010","journal-title":"Am. J. Orthod. Dentofac. Orthop."},{"key":"ref_34","doi-asserted-by":"crossref","unstructured":"Jaramillo-Bedoya, D., Villegas-Giraldo, G., Agudelo-Su\u00e1rez, A.A., and Ram\u00edrez-Ossa, D.M. (2022). A Scoping Review about the Characteristics and Success-Failure Rates of Temporary Anchorage Devices in Orthodontics. Dent. J., 10.","DOI":"10.3390\/dj10050078"},{"key":"ref_35","doi-asserted-by":"crossref","unstructured":"Reznikov, N., Dagdeviren, D., Tamimi, F., Glorieux, F., Rauch, F., and Retrouvey, J.M. (2019). Cone-Beam Computed Tomography of Osteogenesis Imperfecta Types III and IV: Three-Dimensional Evaluation of Craniofacial Features and Upper Airways. J. Bone Miner. Res. Plus, 3.","DOI":"10.1002\/jbm4.10124"},{"key":"ref_36","doi-asserted-by":"crossref","unstructured":"Messineo, D., Luzzi, V., Pepe, F., Celli, L., Turchetti, A., Zambrano, A., Celli, M., Polimeni, A., and Ierardo, G. (2020). New 3D Cone Beam CT Imaging Parameters to Assist the Dentist in Treating Patients with Osteogenesis Imperfecta. Healthcare, 8.","DOI":"10.3390\/healthcare8040546"},{"key":"ref_37","doi-asserted-by":"crossref","unstructured":"Di Carlo, G., Saccucci, M., Ierardo, G., Luzzi, V., Occasi, F., Zicari, A.M., Duse, M., and Polimeni, A. (2017). Rapid Maxillary Expansion and Upper Airway Morphology: A Systematic Review on the Role of Cone Beam Computed Tomography. Biomed. Res. Int., 2017.","DOI":"10.1155\/2017\/5460429"},{"key":"ref_38","doi-asserted-by":"crossref","unstructured":"Uribe, F., Mehr, R., and Mathur, A. (2015). Failure rates of mini-implants placed in the infrazygomatic region. Prog. Orthod., 16.","DOI":"10.1186\/s40510-015-0100-2"},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"1595","DOI":"10.1016\/j.joms.2009.04.015","article-title":"Factors associated with the stability of mini-implants for orthodontic anchorage: A study of 414 samples in Taiwan","volume":"67","author":"Wu","year":"2009","journal-title":"J. Oral Maxillofac. Surg."}],"container-title":["Journal of Clinical Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2077-0383\/14\/1\/55\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T17:00:27Z","timestamp":1760115627000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2077-0383\/14\/1\/55"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,12,26]]},"references-count":39,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2025,1]]}},"alternative-id":["jcm14010055"],"URL":"https:\/\/doi.org\/10.3390\/jcm14010055","relation":{},"ISSN":["2077-0383"],"issn-type":[{"type":"electronic","value":"2077-0383"}],"subject":[],"published":{"date-parts":[[2024,12,26]]}}}