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Table of Contents   
CASE REPORT  
Year : 2011  |  Volume : 22  |  Issue : 3  |  Page : 468-471
Concomitant hypo-hyperdontia with dens invaginatus


1 Department of Oral Pathology and Microbiology, K M Shah Dental College and Hospital, Pipariya, Vadodara, Gujarat, India
2 Department of Oral and Maxillofacial Surgery, S J M Dental College and Hospital, Chitradurga, India
3 Rishiraj College of Dental Sciences and Research Center, Gandhinagar, Bhopal, India

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Date of Submission03-Oct-2009
Date of Decision18-Sep-2010
Date of Acceptance16-Oct-2010
Date of Web Publication3-Nov-2011
 

   Abstract 

Although developmental anomalies of tooth number are quite common in permanent dentition, concomitant occurrence of hypohyperdontia is a very rare mixed numeric anomalous condition of teeth. Very few cases of this condition have been reported in the English literature. Here we report such a rare case noted in a 26 year-old male dental graduate with no other associated systemic condition or syndrome.

Keywords: Dens invaginatus, hypodontia, hyperdontia, missing teeth, supernumerary teeth

How to cite this article:
Manjunatha B S, Nagarajappa D, Singh SK. Concomitant hypo-hyperdontia with dens invaginatus. Indian J Dent Res 2011;22:468-71

How to cite this URL:
Manjunatha B S, Nagarajappa D, Singh SK. Concomitant hypo-hyperdontia with dens invaginatus. Indian J Dent Res [serial online] 2011 [cited 2019 Nov 13];22:468-71. Available from: http://www.ijdr.in/text.asp?2011/22/3/468/87072
Concomitant hypohyperdontia (CHH) is the convenient concise term introduced by Camilleri to describe the simultaneous presence of hypodontia and supernumerary teeth. [1] Etiology is obscure, and as asserted by Baccetti, the two occurrences are probably unrelated phenomena. [2] Disturbance in migration, proliferation and differentiation of the neural crest cells and interaction between the epithelial and mesenchymal cells during initiation stage of tooth development have been suspected as possible cause. [3]

A non-syndromic case of concomitant multiple supernumerary teeth in both arches and missing maxillary permanent left canine in a 12 year-old girl was reported by Sharma. [3] Brooke and Winter and Camilled each had also reported a case of missing upper lateral incisor and presence of mesiodens, while Munns and Nathanail each reported a case of missing upper lateral incisors associated with supplemental premolars. [4]

This case report presents the rare association of two dental anomalies: a supplemental supernumerary tooth with dens invaginatus and congenital absence of bilateral mandibular central incisors. This type of unusual occurrence of CHH with dens invaginatus is rarely reported in the literature.


   Case Report Top


A healthy, 26 year-old male dental graduate presented with a complaint of missing lower front teeth and also an extra tooth in left maxillary anterior area.

Intra-oral clinical examination revealed retained deciduous mandibular central incisors and bilateral absence of permanent mandibular central incisors [Figure 1]. There was presence of an extra tooth in the maxillary anterior area between the lateral incisor and canine, which resembled supplemental supernumerary tooth [Figure 2], with the following teeth present

Maxillary arch: 18,17,16,15,14,13,12,11,21,22,SN,23,24,25,26,27,28

Mandibular arch: 48,47,46,45,44,43,42,81,71,32,33,34,35,36,37,38.
Figure 1: Photograph showing over-retained bilateral deciduous central incisors and absence of permanent central incisors

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Figure 2: Photograph showing supplemental supernumerary lateral incisor

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The supplemental supernumerary tooth also showed dens invaginatus extending till the cervical line on the lingual aspect. Over-retained deciduous mandibular central incisors and congenital absence of both permanent mandibular central incisors and supplemental incisor along with dens invaginatus [Figure 3] were quite evident clinically.
Figure 3: Photograph showing (mirror image) dens invaginatus associated with supplemental tooth

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Radiographic examination using orthopantamogram confirmed the congenital absence of permanent mandibular central incisors bilaterally;and also the supplemental supernumerary tooth distal to maxillary lateral incisor [Figure 4]. Intra-oral periapical (IOPA) radiographs showed dens invaginatus associated with supplemental tooth [Figure 5] and retained deciduous mandibular central incisors without any evidence of root resorption [Figure 6].
Figure 4: Orthopantamogram showing supplemental tooth distal to maxillary lateral incisor (arrow) and over-retained deciduous mandibular central incisors (arrow head)

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Figure 5: IOPA radiograph showing dens invaginatus associated with supplemental tooth

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Figure 6: IOPA radiograph showing retained deciduous mandibular central incisors without any evidence of root resorption

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A final diagnosis of CHH was made on the basis of clinical and radiographic findings.


   Discussion Top


Disturbances in craniofacial development lead to various dental anomalies, including tooth agenesis and supernumerary teeth. Any alterations in number of teeth are the most common anomaly in humans.

Simultaneous occurrence of hypodontia and a supernumerary tooth in an individual is termed as "concomitant hypodontia and hyperdontia" by Camilleri and "hypo-hyperdontia" by Gibson. [5] However, concomitant tooth agenesis (hypodontia) and supernumerary teeth is very rare in the general population. [6] A small number of cases of co-existing hypo-hyperdontia have been reported in the English literature. [1],[3],[4],[5],[6],[7] A recently published report comprising a large sample of 2108 non-syndromic orthodontic patients aged from 7 to 16 years, studied retrospectively, showed 0.33 % of CHH cases. The authors also found a correlation between CHH and some syndromes. [8]

In 1967, Mumms and Camilleri reported first two cases of hypodontia and supernumerary teeth occurring in the maxillary arch. Later, Spyropoulos et al. in 1979 reported three more cases of CHH without any association of other abnormalities and suggested that the routine use of panoramic radiographs is very helpful in the detection of CHH. [5]

In the present report, both medical and dental histories were uneventful. There was no history of trauma to the dentition and jaws during infancy or childhood. In addition, there was no family history of any developmental anomalies of teeth either in patient's parents or siblings. Extra orally, all structures including hairs, eyes and skin appeared normal.

Clinical and radiographic findings were confirmed from the study casts which were made from impressions of upper and lower arches. The alignment of teeth in both arches was almost close to normal without any crowding or other major abnormalities that necessitate orthodontic and/or prosthodontic treatment. The patient expressed satisfaction with his dental appearance and was not ready for any kind of treatment. Hence, no treatment was rendered as he was not interested. The team of clinicians tried convincing for esthetic recontouring of retained deciduous incisors, which he may get it done in the near future. In cases like the one reported here, to ensure a complete esthetic and functional rehabilitation, a multidisciplinary approach comprising a team of orthodontist, oral surgeon and prosthodontist may be adopted.

Several population studies have been carried out in the past to establish the epidemiological, clinical and genetic characteristics and prevalence of hypodontia both in primary and permanent dentition, through the collection of the dental history of individuals belonging to several families representing various countries/geographic locations and races. It is apparent from these studies that the population prevalence of hypodontia varies with geographic location, which ranges from 2.5 to 7.9% in permanent dentition and 0.3 to 2.6% of total population. [9]

Recent studies suggest that both genetic and environmental factors have been found to contribute to the etiology of tooth agenesis and supernumerary teeth. The future identification and analysis of genetic basis is very essential to treat these conditions better. So, the clinician should collaborate and refer a geneticist for further investigative research. [9],[10]

This report describes the unusual non-syndromic case of coexistent hypodontia and supplemental supernumerary tooth with dens invaginatus seen clinically and radiographically. Of interest to the authors was the presence of dens invaginatus associated with supplemental lateral incisor. To the authors' best of knowledge, no such case has been reported describing CHH with dens invaginatus and this rarity prompted to report the case.

Few cases of CHH occurring in an individual have been reported and many are associated with dento-facial irregularities or syndromes. However, in the present case, a supplemental supernumerary tooth in the maxillary arch is within the arch and showed no crowding or any other major esthetic, phonetic and functional irregularities.

To conclude, it is suggested that detailed studies of samples of CHH are required that investigate the prevalence and location of any missing and extra teeth, the size and morphology of remaining teeth in the dentition and also the pattern of tooth size, shape and number in relatives of affected individuals. Such studies may lead to a better understanding of underlying factors that are involved in the pathogenesis.

 
   References Top

1.Camilleri GE. Concomitant hypodontia and hyperodontia. Case report. Br Dent J 1967;123:338-9.   Back to cited text no. 1
[PUBMED]    
2.Baccetti T. A controlled study of associated dental anomalies. Angle Orthod 1998;68:267-74.  Back to cited text no. 2
[PUBMED]  [FULLTEXT]  
3.Sharma A. A rare non syndrome case of concomitant multiple supernumerary teeth and partial anodontia. J Clin Pediatr Dent 2001;25:167-9.   Back to cited text no. 3
[PUBMED]    
4.Low T. Hypodontia and supernumerary tooth: Report of a case and its management. Br J Orthod 1977;4:187-90.  Back to cited text no. 4
[PUBMED]    
5.Spyropoulos ND, Patsakas AJ, Angelpoulos AP. Simultaneous presence of partial anodontia and supernumerary teeth. th Oral Surg Oral Med Oral Pathol 1979;48:53-6.  Back to cited text no. 5
    
6.Bateman G, Mossey PA. Ectopia or concomitant hypohyperdontia? A case report. J Orthod 2006;33:71-7.  Back to cited text no. 6
[PUBMED]  [FULLTEXT]  
7.Segura JJ, Jimenez-Rubio A. Concomitant hypohyperdontia:Simultaneous occurrence of a mesiodens and agenesis of a maxillary lateral incisor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;86:473-5.  Back to cited text no. 7
    
8.Varela M, Arrieta P, Ventureira C. Non-syndromic concomitant hypodontia and supernumerary teeth in an orthodontic population. Eur J Orthod 2009;31:632-7.  Back to cited text no. 8
[PUBMED]  [FULLTEXT]  
9.Pemberton TJ, Das P, Patel PI. Hypodontia: Genetics and future perspectives. Braz J Oral Sci 2005;4:695-706.  Back to cited text no. 9
    
10.Mattheeuws N, Dermaut L, Martens G. Has hypodontia increased in Caucasians during the 20th century? A meta analysis. Eur J Orthod 2004;26:99-103.  Back to cited text no. 10
[PUBMED]  [FULLTEXT]  

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Correspondence Address:
B S Manjunatha
Department of Oral Pathology and Microbiology, K M Shah Dental College and Hospital, Pipariya, Vadodara, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.87072

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]

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