An unusual case of nonsyndromic complete canine agenesis in permanent dentition

Supreet Shirolkar; Khooshbu Gayen; Khushboo Rana; Rajib Sikar; Subir Sarkar

doi:10.4103/ijohr.ijohr_13_21

CASE REPORT

Year : 2021 | Volume : 7 | Issue : 2 | Page : 84-86

An unusual case of nonsyndromic complete canine agenesis in permanent dentition

Supreet Shirolkar, Khooshbu Gayen, Khushboo Rana, Rajib Sikar, Subir Sarkar
Department of Pedodontics and Preventive Dentistry, Dr R Ahmed Dental College and Hospital, Kolkata, West Bengal, India

Date of Submission	19-May-2021
Date of Acceptance	13-Jul-2021
Date of Web Publication	23-Dec-2021

Correspondence Address:
Khooshbu Gayen
Department of Pedodontics and Preventive Dentistry, Dr R Ahmed Dental College and Hospital, Kolkata, West Bengal
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/ijohr.ijohr_13_21

Abstract

Permanent canine agenesis is a rare condition but absence of permanent canines in both the maxillary and mandibular arches are even rarer. It can happen on its own or in combination with other dental issues. There are very few reports of such cases in the literature. This is the third case in the Indian population that has been reported, to the best of our knowledge. It is critical to have a correct diagnosis and treatment strategy in such rare cases. In an 8-year-old male child, we report an unusual case of canine agenesis of both maxillary and mandibular permanent canines, as well as congenital absence of mandibular central incisors and appearance of shovel-shaped incisors.

Keywords: Agenesis, bilateral, congenital, hypodontia, mandibular, maxillary

How to cite this article:
Shirolkar S, Gayen K, Rana K, Sikar R, Sarkar S. An unusual case of nonsyndromic complete canine agenesis in permanent dentition. Indian J Oral Health Res 2021;7:84-6

How to cite this URL:
Shirolkar S, Gayen K, Rana K, Sikar R, Sarkar S. An unusual case of nonsyndromic complete canine agenesis in permanent dentition. Indian J Oral Health Res [serial online] 2021 [cited 2023 Jun 3];7:84-6. Available from: https://www.ijohr.org/text.asp?2021/7/2/84/333377

Introduction

The congenital abnormality in which there is an absence of teeth, known as hypodontia is among the most well-known dental anomalies.^[1] It is characterized as the developmental absence of teeth in either dentition primary or permanent, excluding third molars. In Caucasians, the prevalence of hypodontia in the permanent dentition ranges from 4.5% to 7.4%.^[2] When it comes to tooth agenesis and gender, females have a higher incidence than males. The third molar is the tooth most frequently found missing, followed by mandibular second premolars (3.4%), maxillary lateral incisors (2.2%), maxillary second premolars (2.1%), and mandibular incisors (0.6%).^[3] Congenital absence of canines in the permanent dentition is extremely rare, with reported incidences ranging from 0.18% to 0.45%.^[4] Canine agenesis has been linked to a variety of other dental anomalies, including malocclusion microdontia, retained primary teeth, agenesis of other teeth, and supernumerary teeth.^[5] A more significant etiological factor has been credited with genetic or familial inheritance. The autosomal dominant inheritance pattern is the most common, and regulatory homeobox genes such as MAX1, PAX9, EDA, and AXIN2 have also been related to dental agenesis.^[6] Tooth agenesis has also been associated with different syndromes such as Down syndrome, Rieger syndrome, Witkop syndrome, and hemifacial microsomia.^[7]

The purpose of this article is to present a rare case of congenitally missing permanent canines in both maxilla and mandible which is extremely rare along with agenesis of permanent central incisors in the mandibular region and the presence of shovel-shaped incisors in the maxillary region.

Case Report

An 8-year-old male patient reported to the Department of Pedodontics and preventive dentistry, Dr. R. Ahmed Dental College and Hospital, with a complaint of irregularly placed upper teeth [Figure 1]. Past medical history also revealed that the patient suffered from congenital nephrotic syndrome with frequent relapse nephrotic syndrome, it was his first visit to the dentist. On intraoral examination, the patient has early mixed dentition, with all four erupted permanent molars, permanent maxillary central and lateral incisors, deciduous maxillary and mandibular canines, permanent mandibular lateral incisors, and permanent maxillary first and second premolars, deciduous mandibular first molars. The permanent maxillary canines, mandibular canines, and mandibular central incisors were absent along with the presence of shovel-shaped maxillary incisors which can be seen in upper intraoral photographs [Figure 2]. There was evidence of caries on the proximal surface of the maxillary right canine and mandibular left deciduous first molar. An orthopantomograph was taken to confirm hypodontia in permanent dentition and it was found that there was no evidence of developing permanent maxillary canines, mandibular canines, and mandibular central incisors [Figure 3].

Figure 1: Vestibular view showing rotated central incisors

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Figure 2: (a) Maxillary occlusal view showing retained primary canine along with shovel-shaped incisors, (b) Mandibular occlusal view showing retained primary central incisors

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Figure 3: Orthopantomogram showing the absence of permanent canines tooth buds in both arches along with the absence of mandibular central incisors tooth buds

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Extraoral examination revealed no abnormalities of the hands or legs, skin, hair, or nails [Figure 4]. Conservative management of the carious tooth is to be performed in subsequent appointments, and the patient was instructed for follow-up every 3 months. Patient consent was obtained in the first appointment for publication of the case. Based on the above findings, the case was diagnosed with congenital agenesis of permanent canines in both arches.

Figure 4: Extraoral photographs showing normal appearance of the head, hands, and legs

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Discussion

Congenital absence of canines in the permanent dentition is extremely rare, with Dolder reporting a 0.06% incidence in 1937,^[8] Bergerstrom 0.23% in 1977,^[9] Davis 0.45% in 1987,^[10] and Fukuta 0.18% in 2004.^[4] The cause of such an anomaly is unknown. According to recent research, human tooth agenesis may be caused by a synergistic interaction of the genes Pax9 and Msx1.^[11] Butler's field theory for mammalian teeth revealed that different tooth types have different rates of tooth agenesis; according to this theory, the most mesially located tooth in each morphological class is the most stable, and since the canine is the only representative feature in its developmental region, it is considered the most stable and rarely missing tooth.^[12] This theory was used by Bailit to describe the differences in tooth agenesis in permanent teeth. The absence of corresponding primary predecessors has been found to be strongly linked to permanent tooth agenesis.

The present case demonstrated agenesis of bilateral maxillary and mandibular permanent canines, as well as both mandibular central incisors, despite the presence of clinically present corresponding predecessors. Agenesis of teeth has been shown to have a negative effect on one's appearance, masticatory ability, speech development, and most importantly, emotional upset during adolescence. It also causes dental arch malpositioning, a lack of maxillary and mandibular bone height development, and periodontal damage.^[13] In their study, Haselden et al. reported a higher survival rate of primary canines in the absence of permanent successors.^[14] Overretention of primary canine can be fundamentally useful in cases of severe hypodontia, as the retention of primary canine may impede the resorption of alveolar bone, thereby preserving bone volume and making implants a viable treatment option.^[14] During childhood and puberty, the upper portion of the mandibular symphysis undergoes major growth changes, which are linked to the continuous eruption of lower incisors. As a result, lower incisor agenesis can have an effect on the mandibular symphyseal region's development.^[15]

The treatment strategy must be tailored to each individual case, taking into account the condition of the primary predecessor, the number of missing teeth, occlusion, and patient/parent preferences. To avoid future complications, it is critical to have a correct diagnosis and treatment strategy in such rare cases. Primary predecessors can indeed be extracted early to allow for spontaneous space closure both with and without further orthodontic alignment, or they can be kept and replaced with a suitable restoration once they are lost. At the time of diagnosis, the present case had almost no resorption of primary canines. The retention of primary predecessors has the advantage that, with the increasing use of implants, alveolar resorption can be delayed until the late teens, allowing for maximum implant placement without the need for bone grafting. Each patient must be assessed individually to determine the best treatment plan.

The main goal, in this case, was to preserve as many of the remaining deciduous maxillary canine and mandibular central incisors as possible. It was decided to keep a regular follow-up for every 3 months

Conclusion

The current case showed complete agenesis of all permanent canines, and it is the third case reported in the Indian population, to the best of our knowledge. Since the presence of such a condition necessitates a multidisciplinary approach, the pediatric dentist's role is to manage the child's behavior, promote good oral hygiene, and provide esthetic rehabilitation.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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