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Received: 18 March 2021 | Revised: 7 December 2021 | Accepted: 8 December 2021

DOI: 10.1111/edt.12728

COMPREHENSIVE REVIEW

Odontoma malformation and disturbances of eruption


subsequent to traumatic dental injuries: A literature review and
a case report

Daniela R. Silva | Anna L. Shahinian

Section of Pediatric Dentistry, University


of California Los Angeles School, Los Abstract
Angeles, California, USA
Due to the close proximity of the primary teeth with the developing permanent suc-
Correspondence cessors, several developmental alterations in the permanent successors have been
Anna L. Shahinian, Section of Pediatric
reported as consequences of traumatic dental injuries to the primary dentition. When
Dentistry, University of California Los
Angeles School, Los Angeles, California, they occur at a very young age such trauma could disturb normal tooth development
714 Tiverton, Los Angeles, CA 90095,
and cause rare anomalies, including sequestration of the permanent tooth germ and
USA.
Email: annashahinian@ucla.edu odontoma-­like malformations. A literature review of case reports with a history of
trauma and odontoma formation is discussed in this paper. This paper also includes a
case report of a patient, who was 3-­years and 5-­months old, when she was seen at the
University of California, Los Angeles Children's Dental Center for the first time. The
grandmother reported that the child was dropped from the father's lap when she was
2 weeks old and suffered head trauma and multiple skull fractures.

KEYWORDS
dental trauma, intrusion injury, permanent tooth, primary tooth

1 | I NTRO D U C TI O N had a TDI when 10–­14 months old. Hypoplasia and hypomineral-
ization were most frequently observed when children had TDIs be-
Through infancy and childhood, children are highly susceptible to tween 16 and 56 months. These higher risks are explained by the
traumatic dental injuries (TDI). The detrimental effects to the de- surrounding bone being less calcified, therefore it does not protect
veloping dentition are related to the type of TDI, the direction and the follicle as it does in older children,3 and this young age coincides
severity of the injury, and the age of the child when the injury oc- with the early phases of odontogenesis.
curred.1 According to Andreasen et al., the prevalence of devel- Odontomas constitute 22% of all odontogenic tumors and are
opmental disturbances secondary to dental injuries in the primary the most common type of benign odontogenic tumor among pa-
dentition ranges from 12% to 69%.1 Deviations to developing teeth tients younger than 20 years of age.4 They are a mass of mineralized
include hypomineralization, circular hypoplasia, crown and root di- tissue, similar in structure to a normal tooth composed of enamel,
lacerations, root duplication, partial or complete arrest of root for- dentin, cementum, and pulp tissue.4 According to Satish et al.,4 the
mation, disturbances in eruption, sequestration of permanent tooth World Health Organization (WHO) classifies odontomas as complex
1
germs, and odontoma-­like formations. or compound. Complex odontomas are the calcified dental tissues
The most severe consequences of TDI in the developing perma- that are simply arranged as an irregular mass bearing no morphologic
nent successors happen when the child is younger than 2 years of similarity to rudimentary teeth. Compound odontomas are com-
age. 2 According to Tewari et al., 2 the total sequestrum of the bud of posed of all odontogenic tissues in an orderly pattern that results
permanent teeth is usually associated with TDI in children younger in many teeth-­like structures but without morphologic resemblance
than 10 months, while odontomas were observed in children who to normal teeth.4 In addition, 61% of odontomas in the anterior

© 2022 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

98 | 
wileyonlinelibrary.com/journal/edt Dental Traumatology. 2022;38:98–104.
|

16009657, 2022, 2, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/edt.12728 by Nat Prov Indonesia, Wiley Online Library on [24/01/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
SILVA and SHAHINIAN 99

segment of the jaws are compound odontomas, whereas the major- odontoma malformations and the length of time lapsed from den-
4
ity in the posterior segment are complex composite odontomas. A tal trauma to the diagnosis of an odontoma could likely be the rea-
review of the literature of case reports of odontoma-­like malforma- son why more cases or studies are not reported in the literature.
tions from 2000 to 2020 indicated, although a rare sequelae to TDIs, Bringing awareness to dentists of all of the possible consequences of
those cases were mostly associated with the age at the time of injury TDIs could help identify more cases that can be reported as a result
and its severity. of such trauma.
The wide range of sequelae to the permanent teeth after TDIs
requires the treating dentist to provide anticipatory guidance to
parents, to adhere to long-­term follow-­ups, and to take a multi-­ 3 | C A S E R E P O RT
disciplinary approach in order to diagnose and treat concomitant
complications. A 3-­years 5-­months old Hispanic female patient presented to the
This paper reviews the recent literature and demonstrates that University of California, Los Angeles Children's Dental Center with
trauma prior to the eruption of primary teeth can also cause de- her grandmother (her legal guardian) in March 2017. The chief com-
velopmental disturbances to the developing permanent dentition plaint was “She has yellow teeth and one has not come out yet.”
which are evident later in childhood. The grandmother reported that at 2 weeks old, the child was
dropped from her father's lap and suffered bilateral skull fractures
with extensive intracranial sub-­dural and sub-­arachnoid hemorrhag-
2 | R E V I E W O F TH E LITE R AT U R E ing requiring her to be hospitalized for 2 weeks. During her hospital-
ization, she experienced several seizures per day and was prescribed
A literature review was conducted on case reports that described oxcarbazepine, which she took until she was 2 years old. Upon dis-
odontoma malformations subsequent to trauma in young children. charge, no complications were reported, and the seizures stopped
An online search of studies and case reports published from 2000 at around 2 years of age. At the initial dental visit, the patient was
through 2020 was performed using PubMed. The database was healthy and taking no medications.
searched using the key phrases/words “odontoma malformation,”
“odontoma-­like malformation,” “sequelae to traumatic dental inju- • A clinical examination revealed the absence of tooth #61 and hy-
ries,” and “trauma to primary teeth.” For this literature review, the poplastic teeth #52, 51, and 62.
case reporting (CARE) and preferred reporting items for case reports • An intraoral periapical radiograph revealed malformed #51, im-
(PRICE) guidelines were used to increase the accuracy, transparency, pacted #61, and odontoma-­like presentation around tooth #11
and usefulness of the case reports.5 A review of case reports includ- (Figure 2B).
ing the age/gender, type of injury, and permanent teeth sequelae is • A panoramic radiograph (Figure 2A) and a cone beam CT (Figure 3)
summarized in Table 1.6–­16 revealed a very large odontoma with three separate segments
Odontomas and odontoma-­
like malformations have been re- of multiple malformed tooth fragments in the anterior maxilla.
ported in patients who had a TDI when aged from 2 weeks old (pres- Additionally, one of the odontomas was suspected of being fused
15
ent case report) to 6 years old. Seven of the twelve case reports to tooth #11. Teeth #21 and #22 were also in close proximity to
reviewed for this article concerned females, and the average age at the odontomas, and there was no radiographic evidence of #12
the time of trauma was 17.6 months. Excluding the outlier case report being present. Tooth #61 was ectopically and horizontally posi-
where an odontoma was observed after trauma to a 6-­year-­old, the tioned, malformed, and hypoplastic.
average age at the time of trauma was found to be 12.2 months. All
cases but one did not report an exact age of the dental trauma. Eleven The patient was referred to the University of California, Los
of the twelve cases were reported in the maxillary anterior region, Angeles Oral and Maxillofacial Surgery Clinic, where surgical ex-
and the most common type of injury sustained was intrusion of a pri- traction of the odontoma was completed in the operatory room in
mary tooth (Figure 1) followed by avulsion. Two of the case reports, April 2018. The family was informed that a second surgical proce-
including the present case, had odontoma malformation as a result of dure would likely be necessary to remove any possible remnants.
trauma to the maxillary ridge prior to primary tooth eruption. After the first surgery, it was understood by the oral surgery team
In these case reports, the odontoma malformation was either ini- that all segments of the odontoma were removed.
6,13,15,16
tiated as a new tooth structure from a detached tooth germ During a periodic examination in July 2019 (2 years after the ini-
(mean age of trauma = 32.8 months) or it resulted in the malforma- tial surgery), the grandmother reported that the patient had a new
tion of the entire permanent tooth germ during the early stages of tooth erupting. Clinically, #11 was palpable in the labial vestibule.
odontogenesis7–­12,14 (mean age of trauma = 13.9 months). Radiographic findings revealed an impacted #21, a small odontoma
Despite a thorough search of the literature, the present study or tooth remnant coronal to #21, and possible malformed crowns of
analyzed only published studies of odontoma malformations subse- #11 and #21.
quent to early childhood dental trauma. This was a limitation due to The patient was referred back to the Oral and Maxillofacial
the lack of available data and prior research. The rare occurrence of Surgery Clinic for a second consultation and a new panoramic
| 100

TA B L E 1 Case Reports Summary

Year Author(s) Age Gender Type of injury Odontoma malformation


6
2001 Heloisa de Oliveira et al. 5 months Male Trauma to maxillary ridge, unerupted Odontoma-­like malformation buccal to #51. All
primary teeth permanent maxillary incisors present
2005 Nelson-­Filho et al.7 1 year 8 months Female Intrusion #52, avulsion of #51 Odontoma-­like malformation of #11
2006 Arenas et al.8 1 year 3 months Male Intrusion of #61 Odontoma malformation of #22
9
2008 Shaked et al. 1 year 6 months Female Intrusion of #51 Anterior maxilla at the floor of the nose, odontoma-­like
malformation of #11
2010 Kuchler et al.10 2 years Male Intrusion of #51 Compound odontoma of #12
2010 Lenzi et al.11 10 months Female Intrusion of primary anteriors Odontoma-­like malformation of #12
2011 Ozdemir et al.12 8 months Female Some primary teeth avulsed and Odontoma-­like malformation of #11
intruded
2013 Padmanabhan et al.13 Less than 4 years Male Trauma to left side of mandible Erupted odontoma with extra-­oral sinus above #37. All
permanent mandibular teeth in quadrant present
2014 Rajni Nagpal and Naveen 1 year 4 months Female Intrusion of primary maxillary incisors Odontoma-­like malformation of #11
Manuja14
2014 Samitri et al.15 6 years Female Intrusion of #51 Compound odontoma palatal to #11. All permanent
incisors present.
2019 Amaral-­Freitas et al.16 6 months Male Maxillofacial trauma, unerupted primary Odontoma-­like malformation on palatal of #12 near
teeth floor of the nose. All permanent incisors present.
2020 Present case 2 weeks Female Maxillofacial trauma, unerupted primary Odontoma-­like malformation of #12
teeth
SILVA and SHAHINIAN

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SILVA and SHAHINIAN 101

F I G U R E 1 (A) Sagittal view showing


close proximity of the primary incisor to
the permanent incisor. (B) Intrusion of the
primary incisory leading to disruption of
the permanent tooth germ. (C) Odontoma
formation (Image created by Dr. Sean
McGivern)

Because of the close anatomic relationship with the apices of the


primary incisors, permanent incisors are under increased risk for
development of malformations.1 In this case report, hypoplasia of
primary anterior teeth, disturbances in eruption, and odontoma-­like
malformations developed as a result of the maxillofacial trauma sus-
tained when the child was 2 weeks old. Injuries to developing teeth
after TDIs have a mosaic of clinical and radiographic presentations.
The age of the child, direction of force, and stage of root resorption
are the associated factors that can lead to multiple malformations
(A)
including the rare odontoma.
Permanent tooth development starts during the embryonic and
fetal periods and continues to adolescence. Children and adolescents
thus retain dental lamina epithelial cells in their maxilla and mandi-
ble.16 According to Wang et al.,17 signaling pathways responsible for
initiating new tooth formation have the potential of developing a su-
pernumerary tooth or an odontoma in response to a traumatic injury
in the local environment. In addition, there are cases of odontomas
which appear to be due to the detachment of a portion of a tooth
(B)
germ which may be from the epithelial sheath of Hertwig or from
the enamel organ.18 In a study by Levy,19 a complex odontoma was
F I G U R E 2 (A) Panoramic radiograph taken in 2017 when the produced by inducing trauma to developing first molars in rats. He
patient was 3 years old with a large odontoma (yellow circle) in the also supported that the occurrence of an odontoma can be caused
anterior maxilla. (B) Occlusal radiograph taken at the initial visit
by separation of a small section of partially differentiated epithelial
in 2017, which showed an odontoma-­like malformation around
cells from the developing tooth.
the developing tooth #11 (Arrow). Tooth #51 appears malformed
with horizontally impacted tooth #61. Teeth 52, 51, and 62 appear Andreasen et al.1 described an odontoma malformation of the
hypoplastic entire permanent tooth germ due to intrusive luxation or avulsion of
the primary tooth. He described a vertically directed force through
radiograph and a cone beam CT (Figure 4A,B). The radiology re- the long axis of the deciduous incisor transmits to the permanent
port indicated multiple malformed supernumerary teeth within tooth germ causing extensive damage. According to this theory, the
individual follicles at the palatal aspect of the maxillary alveolar malformation occurs during the early phase of odontogenesis and
bone. Another odontoma was found anterior to the unerupted affects the morphogenetic stages of the ameloblast development of
#21. A second surgical procedure was completed in January 2020 the permanent tooth germ. Severe complications might be expected
to remove the odontoma between the crowns of the developing when at least half or more of the primary crown is intruded and if
#11 and #21. the successor is in an early developmental stage, the risk of compli-
cations becomes higher. 20
These different theories may explain the varying clinical presen-
4 | DISCUSSION tations seen in the case reports summarized in Table 1. For exam-
ple, in the present case, early trauma at 2 weeks of age may have
Any severe trauma during the early stages of odontogenesis can caused the odontomas to form from the permanent tooth germ of
seriously affect the morphogenetic stages of dental development. the #12. In four of the cases, however, all permanent tooth germs
|

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102 SILVA and SHAHINIAN

F I G U R E 3 (A) Three-­dimensional
computed tomography of the patient
at age three. (B) Series of sagittal views
from the CBCT that show proximity to
the nasal floor and fusion to tooth #11
(Yellow box)

were present in the area that the trauma was induced. These cases crown that has mineralized and developed more will be more resil-
could be supported by the theories of Wang et al.,17 Hitchin,18 and ient to trauma.
19
Levy that a separation of cells from the developing tooth germ led Odontomas are infrequently observed, and the etiology still
to the initiation and growth of an odontoma. It is interesting to note remains unclear. It has been suggested that odontomas have been
that the mean age of trauma for the group with permanent tooth associated with trauma to the primary dentition as well as with infec-
germ odontomas was 13.9 months. This is compared to the group tions, hereditary anomalies, odontoblastic hyperactivity, and genetic
where the odontoma was initiated as a new tooth structure and the alterations responsible for tooth development. These benign tumors
average age of trauma was higher at 32.8 months. This supports the are typically asymptomatic but may lead to retention of deciduous
theory of Andreasen et al.1 that earlier stages of odontogenesis can teeth, non-­eruption of permanent teeth, pain, expansion of the cor-
make the tooth germ itself more susceptible to odontoma malfor- tical bone, and tooth displacement. 22 Conservative surgical excision
mation. Initial calcification of the permanent maxillary central and is the treatment of choice for the aforementioned reasons. However,
lateral incisors happens at 3 months and 11 months, respectively. 21 the development of an odontoma, as a result of a traumatic injury
Trauma to an uncalcified structure can lead to more damage to the to the primary dentition, usually occurs at a very young age when
susceptible and unprotected permanent tooth germ, whereas a the maxillary bone height is small. As odontomas may not erupt
|

16009657, 2022, 2, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/edt.12728 by Nat Prov Indonesia, Wiley Online Library on [24/01/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
SILVA and SHAHINIAN 103

F I G U R E 4 (A) Three-­dimensional
computed tomography of the patient at
age five with axial, coronal, and sagittal
views. Yellow arrow shows a remnant of
the odontoma between the developing
teeth #11 and #21. (B) Series of sagittal
views that show remnants of the
odontoma approximating the nasal floor
(arrow)

(A)

(B)

concomitantly with bone growth and adjacent teeth development, traction of the impacted teeth soon after removal of the lesion may
the location could be high in the maxillary bone, thus significantly be necessary, especially if it is not diagnosed and treated early.6
9
complicating surgery. In the present case, the large size of the mass The patient in this case is seen every 6 months and radiograph-
and its proximity to the nasal cavity and the permanent dentition ically monitored for the eruption of her permanent teeth and any
being in development made the surgery more complicated and in- other anomalies. Long-­term management will include esthetic res-
creased the potential for harm to the permanent teeth. In addition, torations and orthodontic treatment if the permanent teeth do not
when odontomas are associated with unerupted teeth, orthodontic erupt or if they erupt in non-­ideal locations.
|

16009657, 2022, 2, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/edt.12728 by Nat Prov Indonesia, Wiley Online Library on [24/01/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
104 SILVA and SHAHINIAN

Pediatric dentists and general dentists providing dental care to 8. Arenas M, Barbería E, Lucavechi T, Maroto M. Severe trauma in the
children have an ultimate responsibility in active surveillance of the primary dentition—­diagnosis and treatment of sequelae in perma-
nent dentition. Dent Traumatol. 2006;22:226–­3 0.
permanent teeth and appropriate referral to an orthodontist and/or
9. Shaked I, Peretz B, Ashkenazi M. Development of odontoma-­like
oral and maxillofacial surgeon to address treatment for potentially malformation in the permanent dentition caused by intrusion of
impacted or unerupted teeth. A patient with a TDI should be mon- primary incisor—­a case report. Dent Traumatol. 2008;24:395–­7.
itored often and treated by a team of dental specialists to detect 10. Küchler EC, da Silva Fidalgo TK, Farinhas JA, de Castro CM.
Developmental dental alterations in permanent teeth after intru-
early complications and to coordinate timely and comprehensive
sion of the predecessors: clinical and microscopic evaluation. Dent
treatment. Traumatol. 2010;26:505–­8.
11. Lenzi MM, Antero S, Monteiro AM, Campos V. Odontoma-­like mal-
C O N FL I C T O F I N T E R E S T formation in permanent dentition due to severe trauma in decidu-
ous dentition-­c ase report. Braz Dent J. 2010;2:31–­5.
The authors declare that there is no conflict of interest.
12. Ozdemir Y, Akin A, Eden E. Management of multiple sequel-
aes in permanent dentition: 3 years follow-­up. Dent Traumatol.
AU T H O R C O N T R I B U T I O N S 2011;27:67–­70.
Daniela R. Silva involved in concepts, design, data acquisition, man- 13. Padmanabhan MY, Pandey RK, Aparna R. Erupted composite
odontoma associated with malformed teeth—­unusual dental ab-
uscript preparation, and manuscript editing. Anna L. Shahinian in-
errations following maxillofacial trauma. Rom J Morphol Embryol.
volved in literature search, data analysis, manuscript preparation, 2013;54:1153–­6.
and manuscript editing. 14. Rajni N, Naveen M. Sequelae of intrusive trauma to the primary
predecessors: odontoma like malformation and enamel hypoplasia.
DATA AVA I L A B I L I T Y S TAT E M E N T Austin J Dent. 2014;1:1005.
15. Samriti B, Harsimrit K, Ravinder NB, Puneet G. An idiosyncratic
Data sharing is not applicable to this article as no new data were cre-
post-­traumatic tetrad: compound odontome, dentigerous cyst, im-
ated or analyzed in this study. paction, and double-­dilaceration. Quintessence Int. 2014;45:885–­9.
16. Amaral de Freitas G, Brasileiro CB, Zarzar PM, Ferreira FM. Rare
ORCID dental developmental disturbance in primary and permanent
teeth following trauma prior to tooth eruption: case report. Dental
Anna L. Shahinian https://orcid.org/0000-0003-0415-9847
Traumatol. 2020;36(1):79–­83.
17. Wang X-­P, O'Connell DJ, Lund JJ, Saadi I, Kuraguchi M, Turbe-­Doan
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