Surgical Management of a Rare Case of Massive Compound Odontome Associated with Missing Primary Tooth

Abstract

Odontomes are considered to be the most common odontogenic tumours of the jaws. They are benign, mixed tumours arising from the
remnants of both odontogenic epithelium and the ectomesenchyme resulting in the deposition of varied propotions of enamel, dentin,
cementum and pulp tissues. As these lesions show deficiency only in structural arrangement, some authors consider odontomes as
hamartomas or tumour like malformations. Though these lesions are more common in children, very few cases have been reported
in less than five years age group. Blood loss is a major issue in paediatric surgery. Careful and safe surgery is the primary goal of the
surgeon. The external carorid artery which is the only feeder of blood to the face and oral cavity can be ligated to control bleeding in
extensive maxillofacial injuries and orofacial malignancies. Herewith, we report a massive odontome in a three and half-year-old child
which was treated by surgical excision along with carotid control to prevent excess bleeding.

Full text

Journal of Clinical and Diagnostic Research. 2017 Apr, Vol-11(4): ZD10-ZD12
1010
DOI: 10.7860/JCDR/2017/25059.9676
Case Report
Dentistry Section
Surgical Management of a Rare Case of
Massive Compound Odontome Associated
with Missing Primary Tooth
CASE REPORT
A three and half-year-old female child reported to the Department of
Oral and Maxillofacial Surgery with a chief complaint of slow growing
asymptomatic swelling on the left side of the face for the past one
year. On examination, a diffuse swelling measuring 3x3 cm extending
anteroposteriorly from the corner of the mouth to 1 cm anterior to
the tragus of the ear and superioinferiorly from infraorbital rim to the
lower border of the mandible was noted. [Table/Fig-1]. The skin over
the swelling was normal, the nasolabial fold was obliterated and the
lower eyelid was pushed upward. On palpation the swelling was non
tender and bony hard in consistency. Intraoral examination revealed
a swelling in the left maxilla extending from canine to the tuberosity.
The maxillary left lateral incisor was missing and the maxillary first and
second primary molars were displaced but present within the tumour
mass.
Orthopantomogram of the jaws showed a conglomerate mass of
mixed radiodensity extending from the distal aspect of 63 to the
maxillary tuberosity obliterating the left maxillary sinus. The upper first
permanent molar was displaced and pushed up to the superior border
of the sinus however the tooth follicles of the premolars were absent
[Table/Fig-2]. A computed tomogram revealed mixed density lesion
seen in the region of left maxilla with foci of calcifications occupying
the maxillary sinus extending to the subcutaneous plane. However,
no breach in cortex was noted in the medial aspect of the lesion.
Impacted left upper first permanent molar was seen within the maxillary
sinus [Table/Fig-3]. With a radiological differential diagnosis of fibrous
dysplasia, odontome and odontoameloblastoma surgical excision
was planned for the patient. Considering the age of the patient, in an
aid to prevent excessive blood loss during the surgery, external carotid
artery control was considered in addition to the excision.
Carotid Control
A shoulder bag was placed and the neck was extended. A 1.5 cm
incision was placed to expose the carotid triangle, subplatysma flap
was elevated and the sternocleidomastoid muscle was raised from
the investing layer of deep cervical fascia. Posterior belly of digastric
and superior belly of omohyoid were identified and raised. Carotid
sheath was exposed to visualise internal jugular vein, vagus nerve and
the common carotid artery. The common carotid artery was traced up
to the bifurcation of internal carotid artery and external carotid artery.
The external carotid artery was identified and confirmed. Superior
thyroid artery and facial artery were identified and traced up to their
main trunk from the external carotid artery. The hypoglossal nerve was
preserved. After establishing clearance of vital structures the external
carotid artery was tied with 3.0 vicryl to arrest bleeding [Table/Fig-4].
Primary Surgery
A Weber–Fergusson’s incision was made to establish maximum
exposure to the tumour. Multiple teeth like structures with soft tissue
were excised in toto [Table/Fig-5]. A surgical obturator was placed
and the wound was closed. Postoperative recovery was uneventful
[Table/Fig-6] and no recurrence was noted one year after surgery.
Histopathology
The excised lesional mass measured 7x7 cm in dimension; hard in
consistency with an irregular surface containing multiple white areas
admixed with reddish brown areas [Table/Fig-7]. The histopathology
sections of the decalcified tumour bits showed multiple areas of
=
Keywords: Children, External carotid artery ligation, Odontogenic tumour
ELENGKUMARAN SANKARAN1, SANGEETHA NARASIMHAN2, KS SABITHA3, PUNIT CHITLANGIA4
ABSTRACT
Odontomes are considered to be the most common odontogenic tumours of the jaws. They are benign, mixed tumours arising from the
remnants of both odontogenic epithelium and the ectomesenchyme resulting in the deposition of varied propotions of enamel, dentin,
cementum and pulp tissues. As these lesions show deficiency only in structural arrangement, some authors consider odontomes as
hamartomas or tumour like malformations. Though these lesions are more common in children, very few cases have been reported
in less than five years age group. Blood loss is a major issue in paediatric surgery. Careful and safe surgery is the primary goal of the
surgeon. The external carorid artery which is the only feeder of blood to the face and oral cavity can be ligated to control bleeding in
extensive maxillofacial injuries and orofacial malignancies. Herewith, we report a massive odontome in a three and half-year-old child
which was treated by surgical excision along with carotid control to prevent excess bleeding.
[Table/Fig-1]: Image showing the patient with a diffuse swelling on the left side of the face. [Table/Fig-2]: Orthopantomogram depicting mixed radiolucent and radio-opaque
lesion of the left maxilla obliterating the maxillary sinus.

www.jcdr.net Elengkumaran Sankaran et al., Compound Odontome Associated with Primary Tooth
Journal of Clinical and Diagnostic Research. 2017 Apr, Vol-11(4): ZD10-ZD12 1111
are classified into compound and complex odontomes. A compound
odontome results when the dental hard tissues deposition is in such
a way that they anatomically resemble the normal tooth structure.
The compound odontomes are in turn classified into three types
namely denticular type, particular type and conglomerate masses.
When the tissues are not arranged in an orderly fashion a complex
odontome results. Some authors also suggest that odontomes are
a type of supernumerary teeth [1,3]. The surgical specimen of our
case showed multiple miniature teeth like structures favoring the
diagnosis of compound odontome which in turn was confirmed by
the histopathological examination of the same.
The differential diagnosis of odontomes includes odontoamelo-
blastoma, ameloblastic fibroma and fibro-odontomas [5]. The
absence of ameloblastic islands and marked fibrous connective
tissue parenchyma in the histopathology ruled out the possible
diagnosis of these lesions.
Blood supply to the head and neck is obtained from the left and right
external carorid artery which is the branch of the common carotid
artery. Carotid control has been practiced to prevent excessive
bleeding during orofacial surgeries. Slooping of external carotid
artery on the left side facilitated reduced blood supply and thus
reduced blood loss during the surgery. This procedure is considered
safe as it does not result in ischemia to the head and neck tissues
[9,10]. Carotid control attempted in a very young patient for the
excision of a massive odontome is the first of its kind and extensive
literature survey has not shown any such attempt in the past for
treating such a lesion.
CONCLUSION
Though odontomes are benign tumours occurring in the second
decade of life, the dentist should be aware that they can occur
in very small children and can grow to very large size. Extensive
surgeries and severe morbidity in such patients can be avoided
by early diagnosis of these lesions. Dental professionals should
be aware that rarely odontomes can erupt into the oral cavity like
normal tooth. Reporting such a case is to throw light to the surgeons
that practicing carotid control during extensive oral and maxillofacial
surgeries could prevent unnecessary blood loss in children.
enamel spaces, dentin, pulp and cemental tissues that resembled
miniature tooth structures [Table/Fig-8].
DISCUSSION
Odontomes are defined as “tumours formed by the overgrowth
of transitory or complete dental tissues” [1]. Odontomes account
to about 22% of all the odontogenic tumours and can either be
solitary or multiple in number [2,3]. The aetiology of these lesions is
unknown. However, trauma, infection and loss in genetic control of
tooth development have been quoted as possible causative factors
[1,2,4]. Their association with syndromes like gardner’s syndrome,
hermann’s syndrome and basal cell nevoid syndrome prove its
genetic predisposition [5].
Odontomes manifest as asymptomatic swellings and are usually
smaller than the size of the normal tooth [2,5]. Our case exhibited
a massive swelling causing the expansion of the maxillary cortex
which is very unusual.
Odontomes are associated with impacted teeth and at times
missing tooth [2]. In our case the primary left lateral incisor and the
permanent premolars were missing. The first permanent molar was
impacted. The probable origin of this lesion could be attributed to the
tooth germs which were missing in this patient. A large odontome
in this case might have resulted due to uncontrolled proliferation
of the terminally differentiated ameloblasts, odontoblasts and
cementoblasts thereby secreting enamel, dentin and cementum,
due to loss of genetic control in the alignment of the these hard
tissues as proposed by Iatrou I et al., [5].
Odontomes are usually intrabony lesions, but occasional cases
in the gingiva have been reported in the literature [5]. Compound
odontomes are common in the anterior maxilla and complex
odontomes are prevalent in the posterior part of the jaws [6].
Occasionally, odontomes erupt into the oral cavity like normal teeth.
So far about 22 cases of erupted odontomes have been reported
in literature [7,8]. Our case was peculiar that it involved almost the
entire left quadrant of the maxilla with multiple teeth like structures
embedded within.
Based on their structural composition and arrangement, odontomes
[Table/Fig-3]: Computed tomographic image of the patient depicting mixed density lesion along with foci of calcifications. [Table/Fig-4] Image showing the ligation of the left
external carotid artery. [Table/Fig-5]: Intraoperative image showing the large tumour mass.
[Table/Fig-6]: Image showing the postoperative extraoral view of the patient after a week. [Table/Fig-7]: Image showing the excised tumour mass consisting of a conglom-
erate of dental hard tissues. [Table/Fig-8]: A and B-Photomicrograph showing the histopathology section of the surgical specimen depicting tooth like structures (H and E
20X).

Elengkumaran Sankaran et al., Compound Odontome Associated with Primary Tooth www.jcdr.net
Journal of Clinical and Diagnostic Research. 2017 Apr, Vol-11(4): ZD10-ZD12
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PARTICULARS OF CONTRIBUTORS:
1. Reader, Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, Sri Ramachandra University, Porur, Tamil Nadu, India.
2. Lecturer, Department of Oral and Craniofacial Health Sciences, College of Dental Medicine, University of Sharjah, Sharjah-U.A.E.
3. Professor and Head, Department of Oncology, Kidwai Memorial Institute of Oncology, Bengaluru, Karnataka, India.
4. Reader, Department of Oral and Maxillofacial Surgery, Mahatma Gandhi Dental College and Hostipal, Jaipur, Rajasthan, India.
NAME, ADDRESS, E-MAIL ID OF THE CORRESPONDING AUTHOR:
Dr. Sangeetha Narasimhan,
M28-211, Department of Oral and Craniofacial Health Sciences, College of Dental Medicine,
University of Sharjah, Sharjah-U.A.E.
E-mail: sangeetha_narashiman@yahoo.co.in
FINANCIAL OR OTHER COMPETING INTERESTS: None.
Date of Submission: Oct 26, 2016
Date of Peer Review: Dec 02, 2016
Date of Acceptance: Jan 13, 2017
Date of Publishing: Apr 01, 2017
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