Three‐dimensional evaluation of morphology and position of impacted supernumerary teeth in cases of cleidocranial dysplasia

Cleidocranial dysplasia (CCD) is a congenital anomaly characterized by the presence of impacted supernumerary teeth and delayed eruption of permanent teeth. However, there has been no detailed investigation on supernumerary teeth in patients with CCD using three‐dimensional (3D) imaging techniques. The purpose of this study was to elucidate the morphology and position of supernumerary teeth using 3D images reconstructed from cone‐beam computed tomography (CBCT) data in a group of five Japanese subjects (male, 3; female, 2; age, 15.0‐25.4 years) with CCD. All five subjects exhibited supernumerary teeth (39 in total; average, 7.8; range, 1‐15). All supernumerary teeth were impacted and existed as pairs with adjacent permanent teeth. Comparison of the size (the crown and dental‐root lengths, the crown mesiodistal and buccolingual diameters), the number of cusps and dental roots, the position, and direction of supernumerary teeth in relation to the adjacent permanent teeth was analyzed. The results of relationship analyses revealed that, at sites other than the molar region, supernumerary teeth were positioned on the lingual and distal sides and supernumerary teeth resembled the morphology of their adjacent permanent teeth in terms of the number of cusps but were smaller than the adjacent permanent teeth. In the molar region, supernumerary teeth were microdontia, which were apparently small and obscure morphologically. In addition, while all adjacent permanent teeth exhibited normal direction, five supernumerary teeth exhibited inverse direction. The findings of this study will improve our understanding of the characteristics of CCD and provide important information for the pathophysiology and clinical treatment.

[1]  J. Clayton-Smith.  Syndromes of the Head and Neck , 1993 .

[2]  Y. Kaneda.  Chronological studies on the formation of the dental roots in the Japanese; morphological studies of the teeth and dentition of the Japanese. , 1956, Okajimas folia anatomica Japonica.

[3]  B. Duncan,et al.  The Gardner syndrome: need for early diagnosis. , 1968, The Journal of pediatrics.

[4]  G. S. Taylor.  Characteristics of supernumerary teeth in the primary and permanent dentition. , 1972, The Dental practitioner and dental record.

[5]  Taylor Gs.  Characteristics of supernumerary teeth in the primary and permanent dentition. , 1972 .

[6]  Primosch Re.  Anterior supernumerary teeth--assessment and surgical intervention in children. , 1981 .

[7]  R. Primosch.  Anterior supernumerary teeth--assessment and surgical intervention in children. , 1981, Pediatric dentistry.

[8]  W. Luckett,et al.  Development of deciduous and permanent dentition in Tarsius and its phylogenetic significance. , 1982, Folia primatologica; international journal of primatology.

[9]  T. Zullo,et al.  Impacted supernumerary teeth: a survey of 50 cases. , 1983, Journal of the American Dental Association.

[10]  S. Kreiborg,et al.  Development of the dentition in cleidocranial dysplasia. , 1990, Journal of oral pathology & medicine : official publication of the International Association of Oral Pathologists and the American Academy of Oral Pathology.

[11]  S. Kreiborg,et al.  Dental treatment strategies in cleidocranial dysplasia , 1992, British Dental Journal.

[12]  I. Thesleff,et al.  Identification of BMP-4 as a signal mediating secondary induction between epithelial and mesenchymal tissues during early tooth development , 1993, Cell.

[13]  F. Hattab,et al.  Supernumerary teeth: report of three cases and review of the literature. , 1994, ASDC journal of dentistry for children.

[14]  S. Kreiborg,et al.  Histological observations of teeth and peridental tissues in cleidocranial dysplasia imply increased activity of odontogenic epithelium and abnormal bone remodeling. , 1995, Journal of craniofacial genetics and developmental biology.

[15]  M. Owen,et al.  Mutations Involving the Transcription Factor CBFA1 Cause Cleidocranial Dysplasia , 1997, Cell.

[16]  S. Kreiborg,et al.  Anomalies of craniofacial skeleton and teeth in cleidocranial dysplasia. , 1999, Journal of craniofacial genetics and developmental biology.

[17]  W. Burgdorf.  Syndromes of the head and neck. 4th ed , 2002 .

[18]  T. Yamashiro,et al.  Expression of Runx1, -2 and -3 during tooth, palate and craniofacial bone development. , 2002, Mechanisms of development.

[19]  T. Yamashiro,et al.  Expression of Runx1, -2 and -3 during tooth, palate and craniofacial bone development , 2002, Mechanisms of Development.

[20]  H. Kanegane,et al.  Functional analysis of RUNX2 mutations in Japanese patients with cleidocranial dysplasia demonstrates novel genotype-phenotype correlations. , 2002, American journal of human genetics.

[21]  L. Rajab,et al.  Supernumerary teeth: review of the literature and a survey of 152 cases. , 2002, International journal of paediatric dentistry.

[22]  D. Rice,et al.  Molecular mechanisms in calvarial bone and suture development, and their relation to craniosynostosis. , 1999, European journal of orthodontics.

[23]  I. Thesleff,et al.  Phenotypic Changes in Dentition of Runx2 Homozygote-null Mutant Mice , 2004, The journal of histochemistry and cytochemistry : official journal of the Histochemistry Society.

[24]  T. Yamashiro,et al.  Runx2 mediates FGF signaling from epithelium to mesenchyme during tooth morphogenesis. , 2004, Developmental biology.

[25]  Sim Heng Ong,et al.  Tooth segmentation of dental study models using range images , 2004, IEEE Transactions on Medical Imaging.

[26]  Y. Fukushima,et al.  Large fontanelles are a shared feature of haploinsufficiency of RUNX2 and its co‐activator CBFB , 2004, Congenital anomalies.

[27]  G. Tell,et al.  Functional analysis of a novel RUNX2 missense mutation found in a family with cleidocranial dysplasia , 2005, Journal of Human Genetics.

[28]  D. Rice.  Craniofacial anomalies: from development to molecular pathogenesis. , 2005, Current molecular medicine.

[29]  Z. Suba,et al.  Cleidocranial dysplasia: diagnostic criteria and combined treatment. , 2005, The Journal of craniofacial surgery.

[30]  Paula Fernández Montenegro,et al.  Retrospective study of 145 supernumerary teeth. , 2006, Medicina oral, patologia oral y cirugia bucal.

[31]  W. Birchmeier,et al.  Continuous tooth generation in mouse is induced by activated epithelial Wnt/β-catenin signaling , 2006, Proceedings of the National Academy of Sciences.

[32]  Paul Montenegro,et al.  Retrospective study of 145 supernumerary teeth. , 2006 .

[33]  A. Banshodani,et al.  A Case Report of Fraternal Twin Sisters who were Diagnosed with Cleidocranial Dysostosis with Delayed Erup tion of the Permanent Teeth , 2007 .

[34]  C. Torii,et al.  Diversity of supernumerary tooth formation in siblings with cleidocranial dysplasia having identical mutation in RUNX2 : possible involvement of non-genetic or epigenetic regulation. , 2007, Orthodontics & craniofacial research.

[35]  S. Oda,et al.  Numerical variation of teeth in the wild house musk shrew Suncus murinus captured from Nagasaki, Japan. , 2008, Archives of oral biology.

[36]  R. Zoroofi,et al.  Rapid Automatic Segmentation and Visualization of Teeth in CT-Scan Data , 2009 .

[37]  H. Liversidge.  Permanent tooth formation as a method of estimating age. , 2009, Frontiers of oral biology.

[38]  T. Sonomura,et al.  Development of deciduous and permanent dentitions in the upper jaw of the house shrew (Suncus murinus). , 2010, Archives of oral biology.

[39]  A. Banshodani,et al.  Correlation between genotype and supernumerary tooth formation in cleidocranial dysplasia. , 2010, Orthodontics & craniofacial research.

[40]  Jiabing Fan,et al.  Molecular genetics of supernumerary tooth formation , 2011, Genesis.

[41]  L. Muscarella,et al.  Identification of a novel RUNX2 gene mutation in an Italian family with cleidocranial dysplasia. , 2011, European journal of orthodontics.

[42]  C. Gurgel,et al.  Cone beam computed tomography for diagnosis and treatment planning of supernumerary teeth. , 2012, General dentistry.

[43]  S. Springate.  The effect of sample size and bias on the reliability of estimates of error: a comparative study of Dahlberg's formula. , 2012, European journal of orthodontics.

[44]  Z. Zhang,et al.  Tooth Lengths of the Permanent Upper Incisors in Patients with Cleft Lip and Palate Determined with Cone Beam Computed Tomography , 2013, The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association.

[45]  Eui-Hwan Hwang,et al.  Identification of supernumerary teeth in 2D and 3D: review of literature and a proposal. , 2013, Journal of dental education.

[46]  N. King,et al.  Prevalence of supernumerary teeth based on panoramic radiographs revisited. , 2013, Pediatric dentistry.

[47]  S. Mallineni,et al.  A comprehensive review of the literature and data analysis on hypo-hyperdontia. , 2014, Journal of oral science.

[48]  M. Omami,et al.  Cone-beam computed tomography exploration and surgical management of palatal, inverted, and impacted mesiodens , 2015, Contemporary clinical dentistry.

[49]  Yeong-Gil Shin,et al.  Fast and Accurate Semiautomatic Segmentation of Individual Teeth from Dental CT Images , 2015, Comput. Math. Methods Medicine.

[50]  K. Moriyama,et al.  Functional tooth restoration utilising split germs through re-regionalisation of the tooth-forming field , 2015, Scientific Reports.

[51]  Nandita Gupta,et al.  Cone-beam computed tomography: An inevitable investigation in cleidocranial dysplasia , 2015, Contemporary clinical dentistry.

[52]  S. Sachdeva,et al.  Inverted and Impacted Maxillary and Mandibular Third Molar: Unusual Case Reports with Review of the Literature , 2015, Saudi journal of medicine & medical sciences.

[53]  M. Lubinsky,et al.  Syndromes with supernumerary teeth , 2016, American journal of medical genetics. Part A.

[54]  T. Ono,et al.  Quantitative analysis of the relationship between maxillary incisors and the incisive canal by cone-beam computed tomography in an adult Japanese population , 2017, Progress in Orthodontics.

[55]  G. Musumeci,et al.  Morphology of palatally displaced canines and adjacent teeth, a 3-D evaluation from cone-beam computed tomographic images , 2018, BMC Oral Health.

[56]  Guanglin Li,et al.  Tooth and Alveolar Bone Segmentation From Dental Computed Tomography Images , 2018, IEEE Journal of Biomedical and Health Informatics.