04 March 2023: Articles
Unknown etiologyMohamad Abdulhamid Alabrash1ABCDEFG*, Mohamed Shaaban Elzouhiry1ABCDEFG, Hend Ahmed AlFadhli2ADE, Ammar Talal Asali2ADEF
Am J Case Rep 2023; 24:e939175
BACKGROUND: Dental anomalies are common congenital disturbances that can occur as single findings or as components of specific syndromes. Primary bi-rooted canine teeth are rare dental anomalies that are more common in the maxilla. It is unusual for a child to have a bi-rooted maxillary canine, as this tooth is known for having a single long root, more than twice the length of the crown. The present report describes the extraction of a bi-rooted primary maxillary canine tooth in a 9-year-old Saudi boy. The report aims to contribute to a better understanding of the possible etiological factors of these rare conditions as well as to review the data available so far in the literature.
CASE REPORT: A 9-year-old Saudi boy presented to the clinic for an initial visit. The patient was medically fit. The chief complaint was “I have pain in my upper front left region”. A thorough oral examination revealed that the upper left primary canine was carious. The panoramic radiograph showed that the former tooth was bi-rooted. The tooth was claimed to be non-restorable. Thus, we planned for extraction. The tooth was extracted in the subsequent visit.
CONCLUSIONS: The presence of bi-rooted primary canines is rare. Dentists should always assess the presence of any dental abnormality. Panoramic radiographs may give an initial sign of the existence of abnormal bi-rooted teeth, and the abnormality can be confirmed by taking intraoral radiographs. Although the data availability in the literature is limited, ethnicity and gender seem to have an impact on its prevalence.
Keywords: Tooth Extraction, Tooth, Deciduous, Child, case reports, Humans, Cuspid, Saudi Arabia, Pain, Radiography, Panoramic, Rare Diseases
Dental anomalies are common congenital disturbances that can occur as single findings or as components of specific syndromes . Gemination, fusion, concrescence, dilaceration, dens in dente, taurodontism, and peg-shaped laterals are examples of developmental anomalies influencing morphology in both deciduous and permanent dentition [1,2]. All these anomalies have clinical implications in terms of aesthetics, malocclusion, and, more importantly, influence on the development of dental decay and oral diseases . There is a link between permanent dentition anomalies and the presence of dental anomalies in the primary teeth, especially when they occur bilaterally . Odontogenesis is a sequential process in which genetic factors play a major role, and alteration of the dental morphology has always been a major concern due to its probable association with general disorders and syndromes . Human beings have 2 sets of teeth: deciduous and permanent. The deciduous (also known as primary) teeth play a key role in a child’s development because they facilitate chewing and speaking, contribute to aesthetics, and protect the integrity of the dental arches, in addition to directing the placement of permanent teeth . The shape, size, number, and composition of the dental tissues of deciduous and permanent teeth can vary significantly. Fewer primary than permanent teeth have abnormal tooth morphology . The most frequently over-retained primary teeth in the oral cavity are the primary upper canines. It is unusual for a child to have a maxillary bi-rooted canine; normally, the primary upper canine has a single long root, more than twice the length of the crown . The present report describes the extraction of a bi-rooted primary maxillary canine tooth in a 9-year-old Saudi boy. The report aims to improve the understanding of the possible etiological factors underlying these rare conditions, and to review the data available so far in the literature.
A 9-year-old Saudi boy visited the dental clinic, department of pediatric dentistry, Batterjee Medical College, Jeddah, Saudi Arabia on the 2nd of November 2021. In the initial visit, a consent form was signed by the guardian (father). The patient’s chief complaint was “I have pain in my upper front left region”. A thorough examination was done on the first visit, and this revealed that intraoral and extraoral soft tissues were within normal limits. Clinically, the patient showed badly decayed primary molars. Based on the clinical examination and patient age, a panoramic radiograph (FONA Art Plus, Milan, Italy) was taken (Figure 1). The panoramic radiograph revealed the presence of a unilateral bi-rooted left maxillary primary canine. This bi-rooted left maxillary primary canine was confirmed by a periapical radiograph (VARIO DG, Dentsply Sirona, Charlotte, NC, USA). Another periapical radiograph, taken with a mesial shift of cone (Figure 2) to apply Clark’s technique, was taken for further confirmation of the presence of 2 roots in the upper left primary canine. A treatment plan was designed based on the clinical and radiographic findings, including extraction of the bi-rooted maxillary left primary canine as the tooth was badly decayed and caries extended to the root with no sufficient remaining walls. Thus, the tooth was non-restorable. The patient was presented to the clinic with a scheduled appointment on the 10th of November 2021 for extraction of the bi-rooted left maxillary primary canine, and the extraction consent was taken and signed by the guardian before the extraction. An oral topical anesthetic gel of benzocaine 20% (Bubble Gum Prime Gel, Prime Dental, Chicago, IL, USA) was applied before the local anesthetic injection to prevent injection discomfort. Then, an injection of mepivacaine hydrochloride 2% with adrenaline 1: 100,000 (Scandonest 2% special, Septodent, Paris, France) was given using a conventional dental aspirating syringe (Septodent, Paris, France) with a 27-gauge, 21-mm short-length dental needle (Nipro, Mechelen, Belgium) as 0.6 ml buccal infiltration and 0.3 ml palatal infiltration. Subjective and objective evaluation of local anesthesia was obtained to ensure the success of pain control. A Molt 9 mucoperiosteal elevator (ZEFFIRO LASCOD, Florence, Italy) was used to reflect the gingival tissues around the tooth. Upper anterior extraction forceps for deciduous teeth (ZEFFIRO LASCOD, Florence, Italy) were used to luxate the tooth, and the tooth was delivered without complications. The extracted tooth is shown in Figures 3 and 4. Copious irrigation with normal saline was followed by gauze packed in the extraction site and the patient was asked to bite for half an hour. Verbal and written postoperative instructions were given and illustrated to the patient and the patient’s guardian. The patient was potentially cooperative, and some behavior guidance techniques - such as distraction - were utilized to reinforce the patient’s positive behavior. Generally, the patient’s behavior could be categorized as positive (+), according to Frankl’s scale .
The patient’s guardian was made aware of the root anomaly and the patient was kept under careful monitoring to ensure the proper eruption of the unerupted permanent successor. The patient had multiple visits to continue his treatment based on the agreed-upon treatment plan, including a lower lingual arch space maintainer. Followup visits were scheduled to take place every 3 months. Patient oral hygiene improvement was noticed in the followup visits.
Bi-rooted primary canines are rare anomalies and might be discovered accidentally during routine screening. Practitioners in the field of dentistry should always be cautious not to overlook any unusual findings, in both deciduous and permanent dentitions. Panoramic radiographs may give an initial sign of the existence of these abnormalities and further indicate the need for an intraoral radiograph to confirm or rule out the presence of such dental anomalies. The detection of this abnormality depends mainly on radiographic examination . The first case reported in the literature for a bi-rooted canine was published in 1941  and the first case report of bi-rooted primary maxillary canines from Saudi Arabia was in 2019 . Two recent case reports – in addition to our case – from Saudi Arabia describe bilateral bi-rooted maxillary canines [9,11]. Our case is the first case of a unilateral bi-rooted primary maxillary canine reported from Saudi Arabia. An important aspect of these case reports to be noted is that, in the 2 recent cases from Saudi Arabia, the occurrence of this anomaly was described in male individuals, and both were 8 to 9 years old at the time of detection [9,11]. A table of reported cases of bi-rooted canines in the literature, which was updated and reviewed by Assiry in 2019, illustrates that, based on these data, this anomaly tends to have a higher incidence in males than in females. Regarding ethnicity, a higher incidence has been reported among Black children (as the author described the ethnicities) .
Treatment alternatives for the affected tooth other than extraction were practically limited as pulpotomy and composite restoration would have a predicted poor prognosis based on the clinical presentation of the affected tooth and high caries risk for this patient.
The limitations of these findings include the nature of this study design itself. A CBCT/Micro-CT would perhaps be a useful image that could have been taken if available, to further dissect the anomaly discussed. It could be important in the future to understand whether or not the condition described in the present report is associated with Sella turcica bridging, as there are some cases of maxillary canine abnormalities and concurrent occurrence of Sella turcica bridging . Maxillary dimensions may also be associated with some dental abnormalities . Future studies are needed on these topics.
Additionally, it could be important in the future to assess whether or not the presence of bi-rooted primary teeth is associated with any other dental anomaly in the primary teeth or permanent successors. Future studies to evaluate the association of dental anomalies with Sella turcica bridging, as well as the relationship with maxillary dimensions, would be beneficial. Furthermore, more studies are needed to assess any association with any facial, skeletal, or systemic conditions.
The presence of a bi-rooted maxillary primary canine is rare. The etiological factors and their association with systemic conditions need further investigation. Although the data availability in the literature is limited, ethnicity and gender seem to have an impact on its prevalence, as it seems to be higher in males compared with females, and is more common in certain racial groups than in others. In general, dentists should always be cautious and look for any abnormalities in the primary and permanent dentitions. Panoramic radiographs may give an initial sign of the existence of these abnormalities and further indicate the need for an intraoral radiograph to confirm or rule out the presence of such dental anomalies. As recent cases of these conditions in the Saudi population were reported, more investigations about causes and factors in the Saudi population may be beneficial.
FiguresFigure 1.. Panoramic radiograph. The panoramic radiograph reveals the presence of a unilateral bi-rooted upper left primary canine. Figure 2.. Periapical radiographs. (A) Periapical radiograph of the upper left primary canine, showing the 2 roots. (B) Periapical radiograph of the upper left primary canine; applying mesial shift confirmed the presence of 2 roots. Figure 3.. The facial aspect of the upper left primary canine after extraction. Figure 4.. The palatal aspect of the upper left primary canine after extraction.
1.. Klein OD, Oberoi S, Huysseune A, Developmental disorders of the dentition: An update: Am J Med Genet C Semin Med Genet, 2013; 163C(4); 318-32
2.. Jahanimoghadam F, Dental anomalies: An update: Adv Hum Biol, 2016; 6(3); 112
3.. Gomes R, Fonseca J, Paula L, Dental anomalies in primary dentition and their corresponding permanent teeth: Clin Oral Investig, 2014; 18(4); 1361-67
4.. Cakan DG, Ulkur F, Taner T, The genetic basis of dental anomalies and its relation to orthodontics: Eur J Dent, 2013; 7(Suppl. 1); S143-S47
5.. King NM, Anthonappa RP, Itthagarun A, The importance of the primary dentition to children – Part 1: Consequences of not treating carious teeth: Hong Kong Practitioner, 2007; 29(2); 52
6.. Talebi M, Parisay I, Khorakian F, Bagherian M, Bi-rooted primary maxillary canines: A case report: J Dent Res Dent Clin Dent Prospects, 2010; 4(3); 101
7.. Phulari RG: Textbook of dental anatomy, physiology & occlusion, 2014; 79-82, Jaypee Brothers Medical Publishers
8.. Dean JA, Avery DR, Mcdonald RE: McDonald and Avery’s dentistry for the child and adolescent, 2022; 346, Elsevier
9.. Almulhim B, Bilateral occurrence of bimaxillary bi-rooted primary canines: A rare case report with review of the literature: Surg Radiol Anat, 2021; 43(6); 997-1000
10.. Takano H, Supernumerary rooted primary mandibular right canine: Shiyo, 1941; 21; 48-50
11.. Assiry A, Bi-rooted primary maxillary canines: A case report: J Med Case Rep, 2019; 13(1); 1-4
12.. Scribante A, Sfondrini MF, Cassani M, Sella turcica bridging and dental anomalies: Is there an association?: Int J Paediatr Dent, 2017; 27(6); 568-73
13.. Woodworth DA, Sinclair PM, Alexander RG, Bilateral congenital absence of maxillary lateral incisors: A craniofacial and dental cast analysis: Am J Orthod, 1985; 87(4); 280-93
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