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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 9  |  Issue : 4  |  Page : 180-182

Nonsyndromic form of oligodontia in a Chinese male patient: A rare case report


1 Department of Oral Medicine and Radiology, Faculty of Dentistry, Melaka-Manipal Medical College, Melaka, Malaysia
2 Department of Pedodontics, Melaka-Manipal Medical College, Melaka, Malaysia
3 Department of Oral Pathology, Melaka-Manipal Medical College, Melaka, Malaysia

Date of Web Publication21-Aug-2017

Correspondence Address:
Preethy Mary Donald
Department of Oral Medicine and Radiology, Faculty of Dentistry, Melaka-Manipal Medical College, Jln Batu Hampar, Bukit Baru 75150, Melaka
Malaysia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jioh.jioh_117_17

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  Abstract 

Oligodontia is one of the developmental disturbances seen in human with more than six congenitally missing teeth. Two forms of oligodontia which exist are the syndromic form and the nonsyndromic form. Mutation or deletion of MSX and PAX genes are implicated in oligodontia. Missing teeth often compromises patient's esthetics, function, and causes mental stress. Here, we highlight a rare case report of a nonsyndromic form of oligodontia with concomitant dental anomalies like taurodontism in maxillary permanent first molar and congenitally missing canine in a 17-year-old apparently normal Chinese male patient.

Keywords: Congenital, missing, permanent


How to cite this article:
Donald PM, Arora A, George R. Nonsyndromic form of oligodontia in a Chinese male patient: A rare case report. J Int Oral Health 2017;9:180-2

How to cite this URL:
Donald PM, Arora A, George R. Nonsyndromic form of oligodontia in a Chinese male patient: A rare case report. J Int Oral Health [serial online] 2017 [cited 2019 Aug 25];9:180-2. Available from: http://www.jioh.org/text.asp?2017/9/4/180/213492


  Introduction Top


Developmental disturbances of fewer than normal complement of teeth can be broadly divided into two categories; hypodontia and oligodontia. Hypodontia is a condition where <six teeth are congenitally missing usually third molars, maxillary lateral incisors, and premolars. Oligodontia is when more than six teeth are congenitally missing excluding third molars. This is often observed during routine intraoral examination procedure by dental professionals. Anodontia is the condition where all the teeth fail to develop which is extremely rare.[1]

The incidence of oligodontia has been found to vary from 0.08% to 0.16%. Oligodontia has been found to occur with or without association of syndrome. Literature has reported many syndromes associated with oligodontia such as ectodermal dysplasia, Seckel syndrome, Down syndrome, Rieger syndrome, Wolf-Hirschhorn syndrome,  Klippel-Feil syndrome More Details, and Van der Woude syndrome.[2],[3],[4],[5]

Among the syndromic form of oligodontia, ectodermal dysplasia is the most common form. The nonsyndromic form of oligodontia (isolated form) lacks the characteristic features of ectodermal dysplasia such as sparse hair, dry skin, dystrophic nails, and deficient sweat glands. Nonsyndromic form can be either familial or sporadic type.[6] For easy understanding to aid in effective case history taking and to differentiate between the two forms clinically, two tables [Table 1] and [Table 2] have been devised: physical and dental traits.[1],[2],[6] Congenitally, missing teeth can be confirmed by taking panoramic radiographs. Here, we present a case report of a nonsyndromic Chinese male patient with oligodontia which includes missing canine which is a rarity.
Table 1: Characteristic features in ectodermal dysplasia

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Table 2: Characteristic features of teeth and dental anomalies associated with syndromic and nonsyndromic form of oligodontia

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  Case Report Top


A 17-year-old Chinese male patient visited our dental clinic, Melaka-Manipal Medical College. He complained of spacing between his upper teeth. The patient also remarked that he is depressed about his smile and appearance. His medical history was unremarkable. As a routine procedure, initial dental screening was done. Extraoral examination revealed normal facial profile. Intraoral examination in relation to maxillary teeth showed midline diastema, clinically missing upper right and left lateral incisors, retained right and left deciduous canine, first molar, and second molar. In relation to mandibular teeth, both right and left deciduous second molar was retained. There were no restorations. Gingiva in relation to lower anteriors had mild inflammation and calculus was evident. A panoramic radiograph was taken and assessment confirmed missing #12, #13, #14, #15, #18, #22, #23, #24, #25 total of 14 congenitally missing teeth including third molars [Figure 1]. Taurodontism was evident with #17 and #27. For further assessment, few more questions were asked. As this was his first dental visit, the patient claimed that neither extractions were done in the past nor remembers an incident of trauma. He also stated that none of his family members had congenital missing teeth. His general examination revealed moderately built and well-nourished condition. His skin and nails were normal. He did not have any intolerance to heat. Based on the given history, clinical features, and radiographic features, it was provisionally diagnosed as nonsyndromic oligodontia.
Figure 1: Panoramic radiograph of a Chinese male patient

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A comprehensive treatment was planned for this patient. Scaling, extraction of all retained deciduous teeth, orthodontic consultation for closure of midline diastema, and prosthetic rehabilitation with removable partial denture were planned. Osseointegrated implants can be planned at a later date for this patient once his skeletal growth of the jaw has been completed. All the treatment procedures were explained to the patient. Scaling was done; however, he did not follow-up for further appointments and hence management of this case came to a halt unfortunately.


  Discussion Top


Oligodontia is a rare developmental disturbance observed in primary or permanent dentition. The underlying cause is due to failure in proliferation of tooth buds from dental lamina.[7] The factors attributed to this condition could be either hereditary or environmental. The condition can be transmitted as autosomal dominant, autosomal recessive, or X-linked. The main genes implicated in early tooth formation are MSX-1 and PAX gene; hence, mutation in any one of the gene can result in agenesis.[8] The environmental factors that can cause oligodontia are mainly due to trauma, infection such as rubella, exposure to drugs such as thalidomide, hormonal influences, previous irradiation, or presence of tumor.[9],[10]

In this case report, there were 14 congenitally missing teeth. However, the patient did not have any other physical characteristics of a syndrome. The teeth missing were maxillary laterals, canine, premolars, mandibular second premolar, and all third molars. Researchers have reported that the most common congenital missing teeth are mandibular second premolars followed by maxillary second premolars and maxillary laterals. However, studies have reported that missing canine is very rare.[8] Interestingly, in our case, both the maxillary canines were congenitally missing which brings the need for reporting this case and to bring awareness of the same among dentists.

Considering the age of patient, it was surprising again that there was no evidence of third molar crown or follicle in all the four quadrants as literature states that crown completion of third molar would be completed by the age of 13 and 14 years for boys and girls, respectively.[11] According to Gupta, it can be termed as severe oligodontia when complete set of molars is missing.[12] Rathore et al. reported nonsyndromic hereditary oligodontia in a 20-year-old female with the absence of ten teeth involving first and second molars and mandibular second premolar.[13] Dental problems concomitant with oligodontia seen in this case were his midline diastema and taurodontism. A significant association between taurodontism and oligodontia had been reported by Seow and Lai.[14] Oligodontia in many patients has been reported to have a strong impact on declining the level of confidence, especially in high schoolchildren and at workplace, due to unsatisfied appearance of smile owing to weak performance. In addition to this, mastication, speech, and malocclusion problems do exist in these kinds of patients, thus deteriorating one's quality of life. Overall, this affects an individual's emotional and psychological balance.[15]

In the present case, a multidisciplinary team of dental professionals consisting of oral surgeon, orthodontists, and prosthodontists is imperative to achieve the goal of restoring esthetics, functional and psychological stress. However, the limitations of this study are that, first, there are no systematic reviews done in relation to this topic to compare, and second, we lost the patient to further complete the treatment procedures as planned.


  Conclusion Top


Reporting of this, rare case will improve further understanding and raise awareness of dental professionals. Dentists while encountering with such patients with oligodontia, a detailed case history, physical examination to rule out syndromic form of oligodontia, and radiographs should be done to diagnose the case. Early systematic intervention will help in improving esthetics, speech, mastication, and social life.

Acknowledgment

The authors would like to express gratitude and appreciation to the radiographer of MMMC, Mrs. Anisah Binti Ismail, for acquisition, exporting, and retrieving image for our publication purpose.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Cawson RA, Odell EW, Stephen R. Porter. Cawson's Essentials of Oral Pathology and Oral Medicine,7 th edn. Edinburgh: Churchill Livingstone;2002.p.19.   Back to cited text no. 1
    
2.
Tangade P, Batra M. Non syndromic oligodontia: Case report. Ethiop J Health Sci 2012;22:219-21.  Back to cited text no. 2
    
3.
De Coster PJ, Verbeeck RM, Holthaus V, Martens LC, Vral A. Seckel syndrome associated with oligodontia, microdontia, enamel hypoplasia, delayed eruption, and dentin dysmineralization: A new variant? J Oral Pathol Med 2006;35:639-41.  Back to cited text no. 3
    
4.
Paradowska A, Szelag J, Slawecki K. Klippel-Feil syndrome – Review of the literature. Dent Med Probl 2007;44:491-4.  Back to cited text no. 4
    
5.
Nieminen P, Kotilainen J, Aalto Y, Knuutila S, Pirinen S, Thesleff I. MSX1 gene is deleted in Wolf-Hirschhorn syndrome patients with oligodontia. J Dent Res 2003;82:1013-7.  Back to cited text no. 5
    
6.
Syed UZ, Chohan AN. Non-syndromic oligodontia of primary and permanent dentition clinical features and early rehabilitation with an esthetic space maintainer in an 8 years old patient. Int Dent J Stud Res 2015;2:36-9.  Back to cited text no. 6
    
7.
Abu-Hussein M, Watted N, Zere E. Nonsyndromic oligodontia in permanent dentition: Three rare cases. IOSR J Dent Med Sci 2015;14:79-83.  Back to cited text no. 7
    
8.
Basoya S, Koduri S, Gupta I, Chandar VV. Familial non-syndromic oligodontia. JIAOMR 2015;27:437-40.  Back to cited text no. 8
    
9.
Prabhuji ML, Ahmed SK, Raghunatha K, Vasudevan V, Murali R, Devaraju D. A rare case of oligodontia. J Indian Acad Oral Med Radiol 2012;24:165-7.  Back to cited text no. 9
  [Full text]  
10.
Jain A, Thakur P, Sarin S. Nonsyndromic oligodontia with ankyloglossia: A rare case report. J Adv Med Dent Sci Res 2015;3:143-7.  Back to cited text no. 10
    
11.
Garn SM, Lewis AB, Bonne B. Third molar formation and its development course. Angle Orthod 1962;32:270-9.  Back to cited text no. 11
    
12.
Gupta M, Panda S, Mutawwam FA, Kariri FM. Diagnosis and management of a patient with congenitally missing maxillary first permanent molars: A rare case report. Case Rep Dent 2016;2016:5891705.  Back to cited text no. 12
    
13.
Rathore R, Deepshika R, Piyush A, Don V, Sumita K. A non-syndromic hereditary oligodontia a rare case report. J Oral Med Oral Surg Oral Pathol Oral Radiol 2016;2:180-3.  Back to cited text no. 13
    
14.
Manjunatha BS, Kovvuru SK. Taurodontism – A review on its etiology, prevalence and clinical considerations. J Clin Exp Dent 2010;2:187-90.  Back to cited text no. 14
    
15.
Rathee M, Mahesh G, Arun K, Richa M, Renu K. Rehabilitation of non-syndromic oligodontia of primary and permanent dentition in young patient – A rare case report. J Interdiscipl Med Dent Sci 2014;2:117.  Back to cited text no. 15
    


    Figures

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    Tables

  [Table 1], [Table 2]



 

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