International Journal of Clinical Pediatric Dentistry

Register      Login

SEARCH WITHIN CONTENT

FIND ARTICLE

Volume / Issue

Online First

Archive
Volume  17 (2024)
Volume  16 (2023)
Volume  15 (2022)
Volume  14 (2021)
Volume  13 (2020)
Volume  12 (2019)
Volume  11 (2018)
Volume  10 (2017)
Volume  9 (2016)
Volume  8 (2015)
Volume  7 (2014)
Volume  6 (2013)
Volume  5 (2012)
Volume  4 (2011)
Volume  3 (2010)
Volume  2 (2009)
Volume  1 (2008)
Related articles

VOLUME 16 , ISSUE 2 ( March-April, 2023 ) > List of Articles

CASE REPORT

A Rare Incidence of Nonsyndromic Mandibular Incisor Agenesis in a Three-generation Family: Case Report and Literature Review

Madhanraj Selvaraj, Karthik Sennimalai, Vilas D Samrit

Keywords : Congenitally missing teeth, Familial tooth agenesis, Hypodontia, Mandibular incisor

Citation Information : Selvaraj M, Sennimalai K, Samrit VD. A Rare Incidence of Nonsyndromic Mandibular Incisor Agenesis in a Three-generation Family: Case Report and Literature Review. Int J Clin Pediatr Dent 2023; 16 (2):388-395.

DOI: 10.5005/jp-journals-10005-2539

License: CC BY-NC 4.0

Published Online: 12-05-2023

Copyright Statement:  Copyright © 2023; The Author(s).


Abstract

Hypodontia is an inherited condition involving the absence of one to six teeth. The permanent dentition is the most frequently affected; however, it may also affect the primary dentition. A congenitally missing tooth (CMT) is the most common dental abnormality, with the missing mandibular second premolar, maxillary lateral incisor, maxillary second premolar, and mandibular central incisor accounting for 90% of CMT in hypodontia studies. The etiology of CMT has been attributed to environmental and genetic contributing factors, with the latter having a strong influence. It may occur in isolation or in association with syndromes. Congenitally missing mandibular incisor is more common in the Asian population and females. Depending on the number and location of missing teeth, hypodontia may be a considerable issue for the clinician since it may impact occlusal balance, mastication, speech, and esthetics and often requires a multidisciplinary approach. Missing mandibular incisors are of particular interest to orthodontists because of the possibility of mandibular retrognathism, the potential for the development of malocclusion, and difficulty in achieving a balanced occlusion. This case report describes the skeletal and dental features of a nonsyndromic familial occurrence of missing mandibular incisors in three generations. A comprehensive literature search was also performed to review the familial cases with missing mandibular incisors.

HTML PDF Share
  1. Kirac D, Eraydin F, Avcilar T, et al. Effects of PAX9 and MSX1 gene variants to hypodontia, tooth size and the type of congenitally missing teeth. Cell Mol Biol (Noisy-le-grand) 2016;62(13):78–84. DOI: 10.14715/cmb/2016.62.13.14
  2. Matalova E, Fleischmannova J, Sharpe PT, et al. Tooth agenesis: from molecular genetics to molecular dentistry. J Dent Res 2008;87(7):617–623. DOI: 10.1177/154405910808700715
  3. De Coster PJ, Marks LA, Martens LC, et al. Dental agenesis: genetic and clinical perspectives. J Oral Pathol Med 2009;38(1):1–17. DOI: 10.1111/j.1600-0714.2008.00699.x
  4. Online Mendelian Inheritance in Man, OMIM®. Johns Hopkins University, Baltimore, MD. MIM Number: #106600: last edited 13/06/2022:https://omim.org/
  5. Polder BJ, Van't Hof MA, Van der Linden FP, et al. A meta-analysis of the prevalence of dental agenesis of permanent teeth. Community Dent Oral Epidemiol 2004;32(3):217–226. DOI: 10.1111/j.1600-0528.2004.00158.x
  6. Khalaf K, Miskelly J, Voge E, et al. Prevalence of hypodontia and associated factors: a systematic review and meta-analysis. J Orthod 2014;41(4):299–316. DOI: 10.1179/1465313314Y.0000000116
  7. Yamaguchi T, Hosomichi K, Yano K, et al. Comprehensive genetic exploration of selective tooth agenesis of mandibular incisors by exome sequencing. Hum Genome Var 2017;4:17005. DOI: 10.1038/hgv.2017.5
  8. Zhang R, Bai Y, Li S. Use of Forsus fatigue-resistant device in a patient with Class I malocclusion and mandibular incisor agenesis. Am J Orthod Dentofacial Orthop 2014;145(6):817–827. DOI: 10.1016/j.ajodo.2013.08.021
  9. Endo T, Yoshino S, Ozoe R, et al. Association of advanced hypodontia and craniofacial morphology in Japanese orthodontic patients. Odontology 2004;92(1):48–53. DOI: 10.1007/s10266-004-0034-5
  10. Rakhshan V, Rakhshan H. Meta-analysis and systematic review of the number of non-syndromic congenitally missing permanent teeth per affected individual and its influencing factors. Eur J Orthod 2016;38(2):170–177. DOI: 10.1093/ejo/cjv008
  11. Rakhshan V. Congenitally missing teeth (hypodontia): a review of the literature concerning the etiology, prevalence, risk factors, patterns and treatment. Dent Res J 2015;12(1):1–13. DOI: 10.4103/1735-3327.150286
  12. Pirinen S, Kentala A, Nieminen P, et al. Recessively inherited lower incisor hypodontia. J Med Genet 2001;38(8):551–556. DOI: 10.1136/jmg.38.8.551
  13. Newman GV, Newman RA. Report of four familial cases with congenitally missing mandibular incisors. Am J Orthod Dentofacial Orthop 1998;114(2):195–207. DOI: 10.1053/od.1998.v114.a87015
  14. Endo T, Ozoe R, Kojima K, et al. Congenitally missing mandibular incisors and mandibular symphysis morphology. Angle Orthod 2007;77(6):1079–1084. DOI: 10.2319/020106-37.1
  15. Goldenberg M, Das P, Messersmith M, et al. Clinical, radiographic, and genetic evaluation of a novel form of autosomal-dominant oligodontia. J Dent Res 2000;79(7):1469–1475. DOI: 10.1177/00220345000790070701
  16. Frazier-Bowers SA, Pham KY, Le EV, et al. A unique form of hypodontia seen in Vietnamese patients: clinical and molecular analysis. J Med Genet 2003;40(6):79e. DOI: 10.1136/jmg.40.6.e79
  17. Korkut E, Gezgin O, Turkoglu S, et al. Bilateral agenesis of permanent mandibular central incisors: two familial case reports. Dent Adv Res 2016;1:105. DOI: 10.29011/2574-7347.100005
  18. Miller MA. An inherited dental anomaly in a Japanese family. J Hered 1941;32(9):313–314. DOI: 10.1093/oxfordjournals.jhered.a105074
  19. Batista MJ, Rihs LB, Sousa Mda L. Risk indicators for tooth loss in adult workers. Braz Oral Res 2012;26(5):390–396. DOI: 10.1590/s1806-83242012000500003
  20. Kaur P, Singh S, Mathur A, et al. Impact of dental disorders and its influence on self esteem levels among adolescents. J Clin Diagn Res 2017;11(4):ZC05–ZC08. DOI: 10.7860/JCDR/2017/23362.9515
  21. Townsend G, Harris EF, Lesot H, et al. Morphogenetic fields within the human dentition: A new, clinically relevant synthesis of an old concept. Arch Oral Biol 2009;54(Suppl 1):S34–S44. DOI: 10.1016/j.archoralbio.2008.06.011
  22. Vastardis H. The genetics of human tooth agenesis: New discoveries for understanding dental anomalies. Am J Orthod Dentofacial Orthop 2000;117(6):650–656.
  23. Thesleff I, Nieminen P. Tooth morphogenesis and cell differentiation. Curr Opin Cell Biol 1996;8(6):844–850. DOI: 10.1016/s0955-0674(96)80086-x
  24. Thesleff I, Vaahtokari A, Vainio S, et al. Molecular mechanisms of cell and tissue interactions during early tooth development. Anat Rec 1996;245(2):151–1561. DOI: 10.1002/(SICI)1097-0185(199606)245:2<151::AID-AR4>3.0.CO;2-#
  25. Fitriasari S, Trainor PA. Diabetes, oxidative stress, and dna damage modulate cranial neural crest cell development and the phenotype variability of craniofacial disorders. Front Cell Dev Biol 2021;9:644410. DOI: 10.3389/fcell.2021.644410
  26. Kılınç G, Bulut G, Ertuğrul F, et al. Long-term Dental Anomalies after Pediatric Cancer Treatment in Children. Turk J Haematol 2019;36(3):155–161. DOI: 10.4274/tjh.galenos.2018.2018.0248
  27. Brook AH. Multilevel complex interactions between genetic, epigenetic and environmental factors in the aetiology of anomalies of dental development. Arch Oral Biol 2009;54(Suppl 1):S3–17. DOI: 10.1016/j.archoralbio.2009.09.005
  28. Gilbert-Barness E. Teratogenic causes of malformations. Ann Clin Lab Sci 2010;40(2):99–114.
  29. Kapadia H, Mues G, D'Souza R. Genes affecting tooth morphogenesis. Orthod Craniofac Res 2007;10(4):237–244. DOI: 10.1111/j.1601-6343.2007.00407.x
  30. Turri de Castro Ribeiro T, Doneux Van der Laan H, Massaro C, et al. Orthodontic treatment of mandibular incisor agenesis with Herbst appliance in a patient with Hanhart syndrome: a 12-year follow-up. Am J Orthod Dentofacial Orthop 2022;161(6):866–877. DOI: 10.1016/j.ajodo.2021.01.035
  31. Ye X, Attaie AB. Genetic basis of nonsyndromic and syndromic tooth agenesis. J Pediatr Genet 2016;5(4):198–208. DOI: 10.1055/s-0036-1592421
  32. Pan Y, Lu T, Peng L, Zeng Q, et al. Functional analysis of ectodysplasin-a mutations in X-linked nonsyndromic hypodontia and possible involvement of X-chromosome inactivation. Stem Cells Int 2021;2021:e7653013. DOI: 10.1155/2021/7653013
  33. Tallón-Walton V, Manzanares-Céspedes MC, Carvalho-Lobato P, et al. Exclusion of PAX9 and MSX1 mutation in six families affected by tooth agenesis. A genetic study and literature review. Med Oral Patol Oral Cir Bucal 2014;19(3):e248. DOI: 10.4317/medoral.19173
  34. Pääkkönen K, Cambiaghi S, Novelli G, et al. The mutation spectrum of the EDA gene in X-linked anhidrotic ectodermal dysplasia. Hum Mutat 2001;17(4):349. DOI: 10.1002/humu.33
  35. Wang J, Sun K, Shen Y, et al. DNA methylation is critical for tooth agenesis: implications for sporadic non-syndromic anodontia and hypodontia. Sci Rep 2016;6:19162. DOI: 10.1038/srep19162
  36. Buschang PH, Julien K, Sachdeva R, et al. Childhood and pubertal growth changes of the human symphysis. Angle Orthod 1992;62(3):203–210. DOI: 10.1043/0003-3219(1992)062<0203:CAPGCO>2.0.CO;2
  37. Chen DD, Cheng JH, Hsu CS. Relationship between craniofacial morphology and congenitally missing mandibular incisors. J Dent Sci 2022;17(2):928–934. DOI: 10.1016/j.jds.2021.12.009
  38. Zhao Z, Zheng L, Huang X, et al. Effects of mouth breathing on facial skeletal development in children: a systematic review and meta-analysis. BMC Oral Health 2021;21(1):108. DOI: 10.1186/s12903-021-01458-7
  39. Aulakh R. The anterior ratio: the missing link between orthodontics and aesthetic dentistry. Case Rep Dent 2013;2013:470637. DOI: 10.1155/2013/470637
  40. Kang HG, Huh YH, Park CJ, et al. Rehabilitation of a patient with non-syndromic partial oligodontia. J Adv Prosthodont 2016;8(3):241–250.
PDF Share
PDF Share

© Jaypee Brothers Medical Publishers (P) LTD.