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Regional odontodysplasia diagnosed and treated via multidisciplinary approach: a case report

  • Dongmei Yin1,2
  • Shaobo Ouyang1,2
  • Peng Zhang1,2
  • Lan Liao1,2,3,4,*,

1Affiliated Stomatological Hospital of Nanchang University, 330006 Nanchang, Jiangxi, China

2The Key Laboratory of Oral Biomedicine of Jiangxi, Jiangxi Province Clinical Research Center for Oral Diseases, 330006 Nanchang, Jiangxi, China

3Clinical Medical Research Center Affiliated Hospital of Jinggangshan University, 343600 Ji'an, Jiangxi, China

4Medical Department of Jinggangshan University, 343600 Ji'an, Jiangxi, China

DOI: 10.22514/jocpd.2023.094 Vol.47,Issue 6,November 2023 pp.191-196

Submitted: 17 October 2022 Accepted: 03 February 2023

Published: 03 November 2023

*Corresponding Author(s): Lan Liao E-mail:


Regional odontodysplasia (RO) is a rare non-hereditary dental anomaly associated with dysplasia. Its etiology remains unclear but is known to affect both the mesodermal and ectodermal dental components, as well as deciduous and permanent dentitions. Its young age of onset and complexity has great physical and psychological impact on the affected patients. However, the clinical management of RO remains unified without standardized treatment guidelines. Thus, this study aimed to report an RO case, the first from Jiangxi Province, China, and discuss its clinical diagnosis and treatment to provide a reference to treat similar cases more effectively in the future.


Odontodysplasia; Ghost teeth; Diagnosis; Therapy; Etiology

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Dongmei Yin,Shaobo Ouyang,Peng Zhang,Lan Liao. Regional odontodysplasia diagnosed and treated via multidisciplinary approach: a case report. Journal of Clinical Pediatric Dentistry. 2023. 47(6);191-196.


[1] Ibrahim Mostafa M, Samir Taha N, Ismail Mehrez MA. Generalised versus regional odontodysplasia: diagnosis, transitional management, and long-term followup—a report of 2 cases. Case Reports in Dentistry. 2013; 2013: 1–5.

[2] Cho S Y. Conservative management of regional odontodysplasia: case report. Journal of the Canadian Dental Association. 2006; 72: 735–8.

[3] Nijakowski K, Wos P, Surdacka A. Regional odontodysplasia: a systematic review of case reports. International Journal of Environmental Research and Public Health. 2022; 19: 1683.

[4] Nijakowski K, Surdacka A. Regional odontodysplasia—a rare developmental dental anomaly. Pediatria Polska. 2021; 96: 153–155.

[5] Sadeghi EM, Ashrafi MH. Regional odontodysplasia: clinical, pathologic, and therapeutic considerations. The Journal of the American Dental Association. 1981; 102: 336–339.

[6] Silva Cunha JL, Barboza Santana AV, Alves da Mota Santana L, Meneses Santos D, de Souza Amorim K, Maciel de Almeida Souza L, et al. Regional odontodysplasia affecting the maxilla. Head and Neck Pathology. 2020; 14: 224–229.

[7] Rushton MA. A new form of dentinal dysplasia: shell teeth. Oral Surgery, Oral Medicine, Oral Pathology. 1954; 7: 543–549.

[8] Zegarelli EV, Kutscher AH, Applebaum E, Archard HO. Odontodysplasia. Oral Surgery, Oral Medicine, Oral Pathology. 1963; 16: 187–193.

[9] Pindborg J J. Pathology of the dental hard tissues. Saunders: Philadelphia. 1970.

[10] Kraus BS, Myers RE, Clark GR. Teratogenic effects of carotid ligation on the developing dentition of the rhesus monkey. Teratology. 1969; 2: 163–172.

[11] Walton JL, Witkop CJ, Walker PO. Odontodysplasia. Oral Surgery, Oral Medicine, Oral Pathology. 1978; 46: 676–684.

[12] Slootweg PJ, Meuwissen PRM. Regional odontodysplasia in epidermal nevus syndrome. Journal of Oral Pathology and Medicine. 1985; 14: 256–262.

[13] Koskinen S, Keski-Filppula R, Alapulli H, Nieminen P, Anttonen V. Familial oligodontia and regional odontodysplasia associated with a PAX9 initiation codon mutation. Clinical Oral Investigations. 2019; 23: 4107–4111.

[14] Gerlach RF, Jorge J, de Almeida OP, Coletta RD, Zaia AA. Regional odontodysplasia. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 1998; 85: 308–313.

[15] Nakatomi M, Morita I, Eto K, Ota MS. Sonic hedgehog signaling is important in tooth root development. Journal of Dental Research. 2006; 85: 427–431.

[16] Dassule HR, Lewis P, Bei M, Maas R, McMahon AP. Sonic hedgehog regulates growth and morphogenesis of the tooth. Development. 2000; 127: 4775–4785.

[17] Hahn H, Wicking C, Zaphiropoulos PG, Gailani MR, Shanley S, Chidambaram A, et al. Mutations of the human homolog of drosophila patched in the nevoid basal cell carcinoma syndrome. Cell. 1996; 85: 841–851.

[18] Johnson R L, Rothman A L, Xie J, Goodrich LV, Bare JW, Bonifas JM, et al. Human homolog of patched, a candidate gene for the basal cell nevus syndrome. Science. 1996; 272: 1668–71.

[19] Bale AE. The hedgehog pathway and basal cell carcinomas. Human Molecular Genetics. 2001; 10: 757–762.

[20] Alotaibi O, Alotaibi G, Alfawaz N. Regional odontodysplasia: an analysis of 161 cases from 1953 to 2017. The Saudi Dental Journal. 2019; 31: 306–310.

[21] HAMDAN MA, SAWAIR FA, RAJAB LD, HAMDAN AM, AL-OMARI IK. Regional odontodysplasia: a review of the literature and report of a case. International Journal of Paediatric Dentistry. 2004; 14: 363–370.

[22] Hovinga J, Ingenhoes R. Regional odontodysplasia. International Journal of Oral Surgery. 1979; 8: 474–477.

[23] Pandis N, Polido C, Bell WH. Regional odontodysplasia. Oral Surgery, Oral Medicine, Oral Pathology. 1991; 72: 492–496.

[24] Ramasetty P, Rai K, Rahim AM, Kappadi D. Regional odontodysplasia: an unusual case report. Journal of Oral and Maxillofacial Pathology. 2009; 13: 62.

[25] Neupert EA, Wright JM. Regional odontodysplasia presenting as a soft tissue swelling. Oral Surgery, Oral Medicine, Oral Pathology. 1989; 67: 193–196.

[26] Crawford PJM, Aldred MJ. Regional odontodysplasia: a bibliography. Journal of Oral Pathology and Medicine. 1989; 18: 251–263.

[27] Gündüz K, Zengin Z, Çelenk P, Özden B, Kurt M, Gunhan O. Regional odontodysplasia of the deciduous and permanent teeth associated with eruption disorders: a case report. 2008; 13: E563–6.

[28] Courson F, Bdeoui F, Danan M, Degrange M, Gogly B. Regional odontodysplasia: expression of matrix metalloproteinases and their natural inhibitors. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 2003; 95: 60–66.

[29] Yang J, Liu Y. Overview of the relationship between cleidocranial dysplasia and abnormal tooth development. Chinese Journal of Practical Stomatology. 2020; 13: 73–7+81.

[30] Zhao ZP, Wang HM, Chen ZY, Meng XJ, Lv BJ. A case of primary failure of tooth eruption and literature review. Journal of Modern Stomatology. 2019; 33: 123–5. (In Chinese)

[31] Dauravu L, Mathew A, Reddy S, Kumar K, Venkataramana V. Ghost teeth: regional odontodysplasia of maxillary first molar associated with eruption disorders in a 10-year-old girl. Journal of Pharmacy and Bioallied Sciences. 2015; 7: 800.

[32] Hess P, Lauridsen E F, Daugaard-Jensen J, Worsaae N, Kofod T, Hermann NV. Treatment strategies for patients with regional odontodysplasia: a presentation of seven new cases and a review of the literature. Oral Health and Preventive Dentistry. 2020; 18: 669–681.

[33] Chinn C, Kohli K. Regional odontodysplasia. A case report. New York State Dental Journal. 2003; 69: 27–9.

[34] Guzman R, Elliott M, Rossie K M. Odontodysplasia in a pediatric patient: literature review and case report. Pediatric Dentistry. 1990; 12: 45–8.

[35] Venkatesh Babu N, Smriti R, Pratima D. Regional odontodysplasia: report of an unusual case involving mandibular arch. Contemporary Clinical Dentistry. 2015; 6: 237–239.

[36] Spini TH, Sargenti-Neto S, Cardoso SV, Souza KCN, de Souza SOM, de Faria PR, et al. Progressive dental development in regional odontodysplasia. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 2007; 104: e40–e45.

[37] Iizawa F, Kinjoh N, Taguchi Y. Regional odontodysplasia: long-term observation of a case on the mandibular left side. Pediatric Dental Journal. 2010; 20: 103–109.

[38] Koruyucu M, Yaman D, Seymen F, Demirel K, Gençay K. Management of regional odontodysplasia: a 10-year-follow-up case report and literature review. European Oral Research. 2018; 52: 111–116.

[39] Ide M, Oshima Y, Chiba T, Adaniya A, Kuroki T, Asada Y. Regional odontodysplasia in maxillary right anterior region—first report: clinical management in relation to 2 case reports. Pediatric Dental Journal. 2020; 30: 124–128.

[40] O’neil D W, Koch M G, Lowe J W. Regional odontodysplasia: report of case. ASDC Journal of Dentistry for Children. 1990; 57: 459–61.

[41] Cahuana A, González Y, Palma C. Clinical management of regional odontodysplasia. Pediatric Dentistry. 2005; 27: 34–9.

[42] Tao SZ, Wang QY, Wu HM, Tang QF, Xu BQ. Three cases of maxillary third molar eruption after total denture restoration. Chinese Journal of Stomatology. 1994; 341. (In Chinese)

[43] Tang Z. Maxillary third molar eruption occurred in 2 cases after maxillary denture restoration. Chinese Medicine. 2001; 266. (In Chinese)

[44] Li X, Cheng M, Sun H, Ouyang J. Advanced research on tooth eruption mechanism. Journal of Jilin University. 2006; 32: 540–542. (In Chinese)

[45] Putra Wigianto AY, Goto T, Iwawaki Y, Ishida Y, Watanabe M, Ichikawa T. Treatment outcomes of implant-assisted removable partial denture with distal extension based on the Kennedy classification and attachment type: a systematic review. International Journal of Implant Dentistry. 2021; 7: 111.

[46] Volpoto L, Botelho G, Casela L, Borges A, Silva K. Regional odontodysplasia: report of a case in the mandible crossing the midline. The Journal of Contemporary Dental Practice. 2008; 9: 142–148.

[47] Abdel‐Kader MA, Abdelazeem AF, Ahmed NEB, Khalil YM, Mostafa MI. Oral rehabilitation of a case with regional odontodysplasia using a regenerative approach—a case report and a review of literature. Special Care in Dentistry. 2019; 39: 330–339.

[48] Ziegler S, Neukam FW. Regional odontodysplasia: orthodontic treatment and transplantation of premolars. American Journal of Orthodontics and Dentofacial Orthopedics. 2012; 142: 710–719.

[49] von Arx T. Autotransplantation for treatment of regional odontodysplasia. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 1998; 85: 304–307.

[50] Canela AHC, Rezende KMPEC, Benitez M, Bönecker M. Early diagnosis of regional odontodysplasia in an infant. Journal of Craniofacial Surgery. 2012; 23: e134–e136.

[51] Dai J, You M, Guo W, Luo S, Shi YC. Regional odontodysplasia: a report of three cases. Chinese Journal of Stomatology. 2020; 55: 501–503. (In Chinese)

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