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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 12  |  Issue : 1  |  Page : 40-44

An unwonted case report of nonsyndromic oligodontia


1 Department of Orthodontics and Dentofacial Orthopaedics, Sardar Patel Post Graduate Institute of Dental and Medical Sciences, Lucknow, Uttar Pradesh, India
2 Department of Orthodontics, Index Institute of Dental Sciences, Indore, Madhya Pradesh, India

Date of Submission17-Jul-2019
Date of Decision21-Sep-2019
Date of Acceptance26-Dec-2019
Date of Web Publication27-Jan-2020

Correspondence Address:
Sumita Jain
Department of Orthodontics and Dentofacial Orthopaedics, Sardar Patel Post Graduate Institute of Dental and Medical Sciences, Lucknow - 226 025, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJDS.IJDS_79_19

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  Abstract 


Dental agenesis is a common developmental anomaly that affects approximately 20% of the population and results in a reduction of the number of teeth present in the oral cavity. Very rarely, more than one anomaly caused due to varying local etiological factors are seen in one patient. The aetiology of dental agenesis may be attributed to either environmental or genetic causes. Environmental factors include, different types of traumas to the alveolar processes, rubella, radiotherapy, use of chemical substances or drugs, as thalidomide and chemotherapy and disturbances in the innervations of the jaw. The absence of teeth is a clinical and public health problem, since the patients in these conditions may present several signs and symptoms as masticatory problems, malocclusion, problems in articulating words, and also the aesthetics may be compromised. These complications may affect self-esteem, behaviour pattern, and social life of these patients. Regarding the diagnosis of oligodontia, it is normally based on radiographic evidence and routine clinical examination, detecting absence of teeth or delayed eruption of them. The panoramic radiography is the most indicated for the diagnosis and study of agenesis, due to this radiographic exam register all maxilla-mandibular regions as well as the development of the tooth germ of the patient with minimal radiation. The motive of this article is to report a case of multiple local etiological factors as the cause of malocclusion and the interceptive management of the case, to prevent aesthetic, functional and psychological problems.

Keywords: Agenesis, nonsyndromic, oligodontia


How to cite this article:
Jain S, Gupta P, Kanungo H. An unwonted case report of nonsyndromic oligodontia. Indian J Dent Sci 2020;12:40-4

How to cite this URL:
Jain S, Gupta P, Kanungo H. An unwonted case report of nonsyndromic oligodontia. Indian J Dent Sci [serial online] 2020 [cited 2020 Aug 13];12:40-4. Available from: http://www.ijds.in/text.asp?2020/12/1/40/276889




  Introduction Top




Agenesis of one or more teeth is one of the most common of human developmental anomalies. Failure of one or more of the third molars to form occurs in 20% of the population.[1],[2] The reported incidence of teeth other than third molars being missing varies from 1.6% to 9.6%. The most commonly missing permanent teeth are the third molars (9%–37%), followed by mandibular second premolars (<3%), maxillary lateral incisors (<2%), and maxillary second premolars and mandibular incisors (<1%).[3]

The dental agenesis is the result of disturbances in the stages of initiation and proliferation during the formation of teeth.[4] Dental agenesis may be manifested as hypodontia, complete anodontia, or as oligodontia.

Very rarely, more than one anomalies caused due to varying local etiological factors are seen in one patient. The aim of this article is to report a case of multiple local etiological factors as the cause of malocclusion including nonsyndromic oligodontia, with agenesis of multiple teeth and disturbances in eruptive pattern. Hence, the interceptive management of such cases becomes the prime concern so as to prevent esthetic, functional, and psychological problems.


  Case Report Top


A father of a 11-year-old girl reported to the clinic with a chief complaint of poor esthetics due to missing teeth in the lower front tooth region. He mentioned, “My daughter did not smile regularly, and when she did it, she put her hand in front of the mouth,” which testified psychological concerns because of her missing teeth. Prenatal and postnatal history as revealed by her father was normal. Past dental and medical history revealed a frenectomy of attached lingual frenum 7–8 months back. The patient was then referred to ear, nose, and throat specialist for speech problem and also was referred to a general physician to rule out any associated syndrome. There was no family history of multiple missing teeth.

The patient was born to noncognate parents with no history of any infection or trauma. The clinical examination revealed mesomorphic built, with an average height-to-weight ratio with normal facial appearance and did not show any physical or skeletal abnormality. The patient was examined to rule out syndromes associated with oligodontia. Radiological examinations did not reveal any significant findings. Other physical examinations also showed no signs of any deformity.

Extraoral examination [Figure 1] and [Figure 2] revealed mesocephalic headform; mesoprosopic, posteriorly divergent face with mild convex profile; acute nasolabial angle and shallow mentolabial sulcus; and incompetent lips with negative lip step. The upper lip was hypotonic with the normal lower lip. Functional examination revealed bilateral synchronous movement of the temporomandibular joint, with bilateral mastication and oronasal respiration.
Figure 1: Extraoral frontal photograph

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Figure 2: Extraoral profile photograph

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Intraoral examination showed multiple missing teeth, a peg-shaped upper left lateral incisor. The patient was caries-free with no history of trauma [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8].
Figure 3: Intraoral frontal occlusion photograph

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Figure 4: Intraoral maxillary view

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Figure 5: Intraoral mandibular view

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Figure 6: Models in occlusion - Right buccal view

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Figure 7: Models in occlusion - Frontal view

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Figure 8: Models in occlusion - Left buccal view

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A panoramic radiograph [Figure 9] was taken which revealed the presence of missing 32, 31, 41, 42, and 12, missing tooth bud in relation to 17, 18, 28, 38, and 48. Furthermore, peg-shaped 21 can be appreciated. Abnormal eruptive pathway can be appreciated in relation to 15 and 35. The crown in relation to 25 was mesially angulated, suggestive of abnormal path of eruption. Lack of space for the eruption of 23 was also appreciated in the orthopantomogram.
Figure 9: Orthopantamogram

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Cephalometric examination [Figure 10] of lateral cephalogram revealed 65%–85% of remaining growth. Skeletal analysis showed Class I skeletal jaw basis with ANB angle of 3° and β angle of 28°. The maxilla was orthognathic, with counterclockwise rotation, suggestive of mild reduction in upper anterior facial height. The mandible was also orthognathic with a clockwise rotation. The Basal plane angle was 35° suggestive of divergent jaw basis. The growth pattern was average toward horizontal, and occlusal plane was tipped downward. The upper incisors were proclined. The upper and lower first molars were intruded. The lower pharyngeal airway space was normal, whereas the upper was reduced.
Figure 10: Lateral cephalogram

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With all the above clinical and radiological findings, diagnosis of nonsyndromic oligodontia with developing skeletal Class I with average toward vertical growth pattern and dentoalveolar angle's Class I malocclusion was made.

Based on the intraoral and radiological findings, the following problems were deduced:

  1. Missing all four lower incisors
  2. Missing upper right lateral incisor
  3. Arrested eruption of upper left canine due to less availability of space
  4. Mesially erupting upper left second molar
  5. Missing right upper second molar tooth bud
  6. Altered eruptive pathway for lower left second premolar and upper right second premolar.


The basic aim was the formulation of treatment plan that will not only solve the esthetic problem but also masticatory problem; to ensure this, a lingual arch with partial denture was planned, in the lower arch. The lingual arch would serve the purpose of holding the molars in place, so that the lower left premolar can get enough space to be guided in proper occlusion. Furthermore, in the upper arch, extraction of mesially erupting upper left second molar was planned, since it had altered eruptive pathway and was also arresting the proper development of the left upper first molar. For guiding both the maxillary canines in occlusion, the treatment in the upper arch was differed for 5–6 months, for the exfoliation of deciduous teeth.


  Discussion Top


The quantum of our understanding is based on post-factum rationale. We are confronted with the clinical picture, and by reasoning backward, we attempt to establish the most likely causative agent. No longer, a clinician can look at a child's mouth, observe missing teeth, and attribute to prolonged retention of deciduous teeth. Yet, in past, local “causes” were stressed in literature as the primary concern. As more knowledge is accumulated in complex behavior of genes, environmental factors and other causative agents, particularly those determining characteristics in our area of endeavor, we are better able to actuate the cause of missing teeth.

Agenesis of one or more teeth is the most common anomaly of dental development in man. Several terms are being used in the literature to describe numeric dental anomalies. One of them, oligodontia literally means “few teeth.” anodontia, an extreme expression of oligodontia, denotes complete absence of teeth. The term “partial anodontia” is frequently used synonymously with oligodontia. Hypodontia is used to indicate a more complex entity, involving not only aberrations in number, size, and shape of the remaining teeth but also abnormalities in the overall rate of dental development and time of eruption.[5],[6]

Permanent dentition is more frequently affected than primary dentition. The incidence for permanent tooth agenesis ranges from 1.6% to 9.6% in the general population excluding third molars.[7],[8],[9],[10] In the primary dentition, tooth agenesis is reported to be 0.5%–0.9%.[11],[12],[13] Severe tooth agenesis (absence of four or more teeth other than third molars) has an estimated prevalence in the general population of 0.25%.[14]

The etiology of dental agenesis may be attributed to either environmental or genetic causes. Environmental factors include different types of traumas to the alveolar processes,[15] rubella,[16] radiotherapy,[17],[18] use of chemical substances or drugs, as thalidomide and chemotherapy,[16] and disturbances in the innervations of the jaw.[19],[20] Based on current knowledge of genes and transcription factors that are involved in the development of teeth, it is assumed that different forms of dental agenesis phenotype observed clinically are caused by mutations in different genes which play distinct roles in the intracellular signaling cascade.[21] This knowledge has led us to the understanding of a wide variety of patterns of agenesis.

Further, agenesis may be either syndromic or nonsyndromic. Various genes involved in nonsyndromic hypodontia in humans include genes encoding transforming growth factor beta and transcription factors (MSX1 and PAX9). These play a critical role during the craniofacial development, as well as genes encoding, a protein involved in Wnt signaling pathway (AXIN2).[21],[22] MSX1 and AXIN2 genes, involved in the early stages of odontogenesis, are associated with tooth agenesis in individuals with disorders such as cleft palate and colorectal cancer.[23] The absence of teeth is a clinical and public health problem,[24] since the patients in these conditions may present several signs and symptoms as masticatory problems, malocclusion, problems in articulating words, and also the esthetics may be compromised. These complications may affect self-esteem, behavior pattern, and social life of these patients.[24],[25] Regarding the diagnosis of oligodontia, it is normally based on radiographic evidence and routine clinical examination, detecting absence of teeth or delayed eruption of them. The panoramic radiography is the most indicated for the diagnosis and study of agenesis; due to this, radiographic examination registers all maxilla–mandibular regions as well as the development of the tooth germ of the patient with minimal radiation. This panoramic radiograph may be preferentially requested as soon as possible, that is, around the 3½ years old.[26],[27] Furthermore, three-dimensional (3D) modalities such as 3D dental volumetric tomography and cone-beam computed tomography can also give us in-depth knowledge about the associated malocclusion.

With the abovementioned data and knowledge, a treatment plan was formulated to maintain the remaining teeth, restore the masticatory functions and improve esthetics, and reassure proper speech. A prosthetic removable partial denture was planned with the objectives to replace the missing teeth, which will restore the masticatory functions, maintaining the position of adjacent natural teeth; preventing undesirable movements such as inclination, extrusion, or migration; improving esthetics; avoiding social problems to the patient, especially in adolescence; and replacing the missing teeth without interfering in the growth of the mandible and maxilla. The replacement of missing teeth with a prosthetic appliance will re-establish the emotional and psychological well-being of the girl and will help her gaining back her self-confidence and smile back.


  Conclusion Top


From the comprehensive history and detailed clinical examination, the case was diagnosed as a nonsyndromic oligodontia. Vigilant treatment planning and thorough insight of conditions such as multiple tooth agenesis is important, so as to rehabilitate the oral environment and establish the affected masticatory and speech functions. Furthermore, the prime importance is to avoid the long-term untoward insinuations. Hence, multidisciplinary treatment planning that involves confirmed and effluent methods is needed to be involved. Patients with tooth agenesis may not only have functional or esthetic problems but also have psychological visitation that requires early diagnosis and competent evaluation for improved oral health overall development of the child.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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2.
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Townsend GC, Brown T. Heritability of permanent tooth size. Am J Phys Anthropol 1978;49:497-504.  Back to cited text no. 6
    
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Eidelman E, Chosack A, Rosenzweig KA. Hypodontia: Prevalence amongst Jewish populations of different origin. Am J Phys Anthropol 1973;39:129-33.  Back to cited text no. 9
    
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Bergström K. An orthopantomographic study of hypodontia, supernumeraries and other anomalies in school children between the ages of 8-9 years. An epidemiological study. Swed Dent J 1977;1:145-57.  Back to cited text no. 10
    
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Grahnen HJ, Granath LE. Numerical variations in primary dentition and their correlation with the permanent dentition. Odont Rev 1961;12:348-57.  Back to cited text no. 11
    
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Ravn JJ. Aplasia, supernumerary teeth and fused teeth in the primary dentition. An epidemiologic study. Scand J Dent Res 1971;79:1-6.  Back to cited text no. 12
    
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Järvinen S, Lehtinen L. Supernumerary and congenitally missing primary teeth in Finnish children. An epidemiologic study. Acta Odontol Scand 1981;39:83-6.  Back to cited text no. 13
    
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Sarnäs KV, Rune B. The facial profile in advanced hypodontia: A mixed longitudinal study of 141 children. Eur J Orthod 1983;5:133-43.  Back to cited text no. 14
    
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Schalk-van der Weide Y, Steen WH, Bosman F. Distribution of missing teeth and tooth morphology in patients with oligodontia. ASDC J Dent Child 1992;59:133-40.  Back to cited text no. 15
    
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Gullikson JS. Tooth morphology in rubella syndrome children. ASDC J Dent Child 1975;42:479-82.  Back to cited text no. 16
    
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Maguire A, Craft AW, Evans RG, Amineddine H, Kernahan J, Macleod RI, et al. The long-term effects of treatment on the dental condition of children surviving malignant disease. Cancer 1987;60:2570-5.  Back to cited text no. 17
    
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Näsman M, Forsberg CM, Dahllöf G. Long-term dental development in children after treatment for malignant disease. Eur J Orthod 1997;19:151-9.  Back to cited text no. 18
    
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Kjaer I, Kocsis G, Nodal M, Christensen LR. Aetiological aspects of mandibular tooth agenesis—focusing on the role of nerve, oral mucosa, and supporting tissues. Eur J Orthod 1994;16:371-5.  Back to cited text no. 19
    
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Andersen E, Skovgaard LT, Poulsen S, Kjaer I. The influence of jaw innervation on the dental maturation pattern in the mandible. Orthod Craniofac Res 2004;7:211-5.  Back to cited text no. 20
    
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De Coster PJ, Marks LA, Martens LC, Huysseune A. Dental agenesis: Genetic and clinical perspectives. J Oral Pathol Med 2009;38:1-7.  Back to cited text no. 22
    
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Agarwal P, Vinuth DP, Dube G, Dube P. Nonsyndromic tooth agenesis patterns and associated developmental dental anomalies: A literature review with radiographic illustrations. Minerva Stomatol 2013;62:31-41.  Back to cited text no. 24
    
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26.
Pilo R, Kaffe I, Amir E, Sarnat H. Diagnosis of developmental dental anomalies using panoramic radiographs. ASDC J Dent Child 1987;54:267-72.  Back to cited text no. 26
    
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10]



 

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