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Case Report

Dentigerous cyst associated with a maxillary permanent

lateral incisor: Case report and literature review
B.K. Ramnarayan, M. Manjunath
Department of Oral Medicine and Radiology, V. S. Dental College and Hospital, K. R. Road, V. V. Puram, Bangalore, Karnataka, India

ABSTRACT
Trauma to deciduous teeth can have severe consequences. Dentigerous cysts are common developmental odontogenic cysts of the
jaws. They are associated with the crown of an unerupted/impacted or developing tooth. Reported cases most commonly involve
mandibular third molars and maxillary canines. They rarely involve the incisors. The condition occurs predominantly in the second
and third decades of life. We report a case of dentigerous cyst involving the permanent maxillary lateral incisor, which developed as
a consequence to trauma to the deciduous predecessor. The pathogenesis and clinical and radiologic features are discussed.

Key words: Cyst, dentigerous cyst, impacted teeth, lateral incisor, unerupted teeth
DOI: 10.4103/0972-1363.52829
DOI

INTRODUCTION dentigerous or follicular cyst as an epithelial developmental

Dentigerous simply means ‘containing teeth’.[1] Dentigerous
cysts are most common benign odontogenic cysts of
developmental type that are usually single in occurrence.
However multiple dentigerous cysts are also reported. [2]
They are generally associated with an impacted tooth
and develop after the complete formation of the crown.[3]
They most commonly involve the mandibular third molars
or the maxillary canine, followed by the mandibular
premolars. The involvement of incisors is rare. Males are
more commonly affected than females. The condition may
occur at any age, but the greatest incidence is reported in
the second and third decades of life.
A dentigerous cyst is one which encloses the crown of
an unerupted tooth by expansion of its follicle and is
attached to the neck. It is important that this definition
is strictly applied and the diagnosis of a dentigerous cyst
is not made uncritically on radiographic evidence alone;
otherwise, keratocyst of the envelopmental variety, follicular
keratocyst and unilocular ameloblastoma involving the
adjacent unerupted teeth are liable to get misdiagnosed.[4]
The classification by the World Health Organization of
epithelial cysts related to odontogenic apparatus refers to the
Address for correspondence:
correspondence Dr. B. K. Ramnarayan, Department of
Oral Medicine and Radiology, Dayananda Sagar college of dental sciences,
Kumarswamy Layout, Bangalore-560078, India.
E-mail: bkramnarayan@hotmail.com
odontogenic cyst.[5]
The diagnostic hypothesis of a dentigerous cyst depends
on a correct radiological examination of the space between
the dental crown and the folliculus, as well as on the
identification of such a space during surgery.[6] The final
diagnosis is possible after histopathological examination.
This article reports a case of a dentigerous cyst in an 8-year-old
boy, involving an unerupted maxillary lateral incisor.
CASE REPORT
A young boy aged about eight years presented with a
chief complaint (as told by the child’s father) of swelling
in the left upper front region of the face, of two months’
duration. It was initially small, but gradually increased in
size. The swelling was associated with occasional dull pain
(on pressure).
There was history of trauma, i.e. he had had a fall two
years ago. He had bleeding from the upper front teeth
region. Four months later the deciduous tooth (61)
exfoliated. The permanent tooth which erupted six months
later was displaced. The patient also gave a history of
difficulty in breathing, since the swelling developed.
General examination showed the boy to be apparently

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Ramnarayan and Manjunath: Dentigerous cyst
healthy with normal growth and development for his age.
He had gross asymmetry of the face due to the swelling on
the left side. Single left submandibular lymph node, oval
in shape measuring about 1.5 cm, was palpable, mobile
and non tender. Extra oral examination showed a diffuse
swelling measuring about 3 × 4 cm, extending from the
midline and left lateral wall of the nose, mildly obliterating
the naso-labial fold and philtrum of the lip to about 1 cm
inferior to the infraorbital margin and outer canthus of
the eye. Mild obliteration of left nares was observed, as
also incompetent lips. Swelling was mildly tender, hard in
consistency [Figure 1].
Intra-oral examination revealed a well-defined swelling,
both palatal and buccal, to 21, 62, 63, 64, with expansion of
cortical pates. Buccally, the swelling measured about 5 × 3
cms, obliterated the labial vestibule and raised the upper lip.
The overlying mucosa showed a bluish discoloration in the
region of 62. Palatally, the swelling extended to the midline
of the palate and measured 2.5 × 3 cms. Swelling was hard
in consistency, except with respect to 62 on the buccal aspect,
where it was soft. The swelling was mildly tender, margins were
well defined. There were no visible or palpable pulsations. 62,
63 were mobile. Associated teeth were non tender. Midline
diastema, extruded and distally displaced 21 was present.
There was spacing between 21, 62, and 63. Eruption was
observed at 12; however 22 had not erupted at the time of
examination [Figures 2 and 3].
Based on the history and clinical examination, a
provisional diagnosis of radicular cyst involving 62, 63
was established. Differential diagnosis of dentigerous cyst
involving 22, adenomatoid odontogenic tumor, unicystic
ameloblastoma, traumatic bone cyst, aneurysmal bone
cyst, calcifying odontogenic cyst, and odontogenic myxoma
were considered.
Radiographs were advised. IOPA and Occlusal radiograph
[Figures 4 and 5] showed an unerupted 22. Unilocular
well defined radiolucency involving the crown of 22 was
noted. Mesially displaced and extruded 21 was found.
The panoramic radiograph showed the developing tooth
bud of 23 to be superiorly displaced to near the floor of
the orbit. Routine blood investigations were within normal
limits. Aspiration of the lesion yielded a straw colored
cystic fluid. Cytologic examination showed needle shaped
cholesterol crystals. Under Hematoxyllin and Eosin (H&E)
smear, inflammatory cells and RBCs were seen. Under
general anesthesia, complete enucleation of the cyst was
done with extraction of 62 and displaced 23 [Figures 6 and
7]. Considering the patient’s age and the fact that it had a
favorable path of eruption, 22 was retained.
142 Journal of Indian Academy of Oral Medicine and Radiology / October - December 2008 / Volume 20 / Issue 4
The enucleated specimen was sent for histopathological
examination, which under H/E stain showed 2-3 layers of
flattened epithelium resembling reduced enamel epithelium.
Connective tissue showed dense collagen fibers and dense
inflammatory infiltrate of predominantly lymphocytes and
plasma cells. Areas of hemorrhage were also seen in the
connective tissue [Figure 8].
Correlating the clinical, radiological and histopathological
features, a final diagnosis of dentigerous cyst involving 22
was established. Follow-up after a month showed that the
swelling had resolved and healing was satisfactory, with
erupting 22 [Figure 9]. Panoramic radiograph showed
formation of bone with trabeculation, suggestive of bony
healing [Figure 10]. Further orthodontic management was
planned for the displaced and extruded teeth.
DISCUSSION
Dentigerous cyst is a cyst arising by separation of the
follicle from around the anatomical crown of an unerupted
tooth within the jaws.[4] It is the second most common
odontogenic cyst[2,7] and represents 33% of all odontogenic
cysts.[3] Its frequency in the general population has been
estimated at 1.44 cysts for every 100 unerupted teeth.[7]
Though present in the first decade of life, it is more common
in the second and third decades of life.[2,4,6] It has a slight
male prediction.[4,6] Studies by Shear showed that 62% of
the subjects studied were males and 38% females, with a
ratio of 1.6: 1.[2] Whites were more commonly affected than
blacks, with a ratio 4.7:1.[2]
Cysts involve impacted, unerupted permanent teeth,
supernumerary teeth, odontomes and, rarely, deciduous teeth.[2]
Seventy five percent of the cases are located in the mandible.[2]
They are most commonly reported in the mandibular third
molar, maxillary canine, mandibular premolar and maxillary
third molar, in that order. The involvement of incisors is very
rare.[3,4] Studies by Shear[2] have shown an incidence of
1.6% involving the lateral incisors, as compared to 45.9%
involving the mandibular third molars. A study by Mourshed
has reported that 1.44% of impacted teeth may undergo
dentigerous cyst transformation.[4] Hence, the involvement
of lateral incisors is rare. Studies by Daley and Wysocki have
reported 0.1%, and by Bernick 2.1%.[6]
Pathogenesis is still controversial. Various hypotheses
have been suggested. Shear[4] advocates an Intra follicular
theory in the development of dentigerous cyst, wherein
he presumes that it starts with the accumulation of fluid
between the reduced enamel epithelium and the enamel,
or between the layers of reduced enamel epithelium and
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Ramnarayan and Manjunath: Dentigerous cyst

Figure 1: Extra-oral photograph showing swelling on the left side of Figure 2: Showing buccal swelling
the face

Figure 3: Showing the palatal swelling Figure 4: IOPA radiograph showing impacted lateral incisor with well
defined radiolucency

Figure 5: Occlusal radiograph showing impacted lateral incisor with Figure 6: Surgical enucleation of the cyst
well defined radiolucency

Figure 7: Gross specimen Figure 8: Histological picture – H&E stain under scanner (x4)
magnification

Figure 9: Post surgical photograph Figure 10: Post treatment panoramic radiograph

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Ramnarayan and Manjunath: Dentigerous cyst
enamel. Al-Talabani and Smith suggested two types of
dentigerous cyst, with different causes arising at different
stages of tooth development. 1) Degeneration of the
stellate reticulum at an early stage of development,
associated with enamel hypoplasia. 2) Development after
crown completion by accumulation of fluid between layers
of reduced enamel epithelium (REE), hypoplasia is not
common.[4]
Another theory advocated by Benn and Main in their study
is that inflammation at the apex of a deciduous tooth can
lead to the development of an inflammatory follicular
cyst around the permanent teeth.[5] Toller postulated that
follicular proliferation occurs, which would eventually lead
to impaction; however, the induction of proliferation is not
known. Killian et al., postulate that trauma to the deciduous
teeth leads to disturbances in the odontogenesis of the
permanent teeth. This can usually give rise to hypoplastic
defects, crown/root disruptions in permanent teeth and
deviation from (N) eruption direction.
The case reported also has a history of trauma to the
deciduous incisors. This could have caused proliferation of
REE and the permanent tooth enamel to develop the cyst.[3]
Another concept suggested by Shear and Lustmann is that
the crown of the permanent teeth erupts into a radicular cyst
of deciduous teeth. This phenomenon may occur, but is rare.
A drawback is that such an erupting tooth may indent rather
than penetrate.[5] The cyst develops between the REE and the
crown of an unerupted tooth in certain cyst-prone individuals,
and it is likely that a genetic factor contributes to the process;
this is yet to be demonstrated at a molecular level.[5]
Regarding the development of cyst, Main (1970) suggested
that pressure exerted by a potentially erupting tooth on an
impacted follicle obstructs the venous outflow and thereby
induces transudation of serum across capillary walls.
Increase in hydrostatic pressure of this pooling fluid results
in separation of follicle from the crown with or without
REE. Capillary permeability is altered so as to permit the
passage of greater percentage of protein. Immunoglobulins
and immunoglobulin containing cells in the cyst wall may
also play a role in fluid formation.[4]
Growth of cyst, presence of glycosaminoglycans (hyaluronic
acid, heparin, chondroitin-4 sulphate) in the walls and
fluids increase the osmolality of cyst fluid which leads to
an increase in internal hydrostatic pressure which results
in cyst expansion. Recent studies have shown presence of
potent bone resorbing factors Prostaglandin E2 (PGE2) and
Prostaglandin E3 (PGE3) bring about bone resorption and
cyst expansion.[4]
144 Journal of Indian Academy of Oral Medicine and Radiology / October - December 2008 / Volume 20 / Issue 4
Clinically, a small dentigerous cyst may be asymptomatic
and may be discovered on routine examination. However,
it can increase to potential size by expansion of bone and,
rarely, bone destruction. Large cysts cause asymmetry of the
face. Displacement of impacted teeth, adjacent teeth and
root resorption is common.[3] Maxillary cysts may displace
and obliterate maxillary antrum and nasal cavity.[2]
On a radiograph, it typically presents as a unilocular
radiolucent area associated with the crown of unerupted
teeth. [3,8] It is usually well-defined with sclerotic borders,
though not always - an infected cyst may show ill-defined
borders.[8] Radiographically, follicular space >2.5 mm is
suggestive of fluid gathering.[3] Radiolucencies greater than
4 mm are to be considered cystic, until otherwise proven.[9]
Depending on the crown–cyst relation, three variants have
been described. 1) In the central variety the cyst surrounds
the crown symmetrically and the crown projects into the
cyst. The tooth may be pushed away from its direction of
eruption. 2) The lateral variety is usually associated with
mesio-angular impacted molars that have partially erupted
and there is dilation of follicle on one aspect of the crown. (3)
In the circumferential variety, the cyst surrounds the crown and
extends for some distance along the root, so that a significant
portion of the root appears to lie within the cyst. It has to be
differentiated from the envelopmental type of keratocyst.[8]
In cases of extensive bony involvement and presence of a
complex cystic lesion, a CT (computerized tomography)
becomes necessary. It helps to rule out solid and fibro-
osseous lesions, displays bony detail and gives exact
information about the size, origin, content and relationship
of the lesion with adjacent structures. An MRI (magnetic
resonance imaging) may fail to show the bony detail but
will precisely display the lesional contents and provide
information about the cyst fluid. The cystic lesion appears
homogeneously hypointense on T1-weighted images and
hyperintense on T2-weighted images.[2]
Microscopic examination of the cyst shows a thin fibrous
cyst wall, epithelial lining of 2-4 cell layers of flattened
non-keratinizing cells - and epithelium-connective tissue
interface is flat. Focal areas of the mucous cells may be
seen in the epithelial lining; capsule/fibrous wall shows
inflammatory cell infiltrate.[8]
Treatment basically consists of enucleation with removal
of unerupted tooth. Enucleation of cyst with retainment of
the involved teeth after the lining is carefully dissected is
done in young individuals, where the eruption is favorable;
marsupialization is done in case of large cyst.[2,3,8] Cysts rarely
recur, except when the lining is not completely removed. [2]
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Ramnarayan and Manjunath: Dentigerous cyst
Metaplastic and dysplastic changes may occur. An
ameloblastoma, mucoepidermoid carcinoma or squamous
cell carcinoma may develop from the lining epithelium of
the cyst.[2,10] Carcinoma arising from a dentigerous cyst is
extremely rare. Gardner has put forward three criteria for
the diagnosis of carcinoma arising from a dentigerous cyst:
1) a microscopic transition area from benign cystic epithelial
lining to invasive malignant squamous cell carcinoma, 2) no
carcinomatous change(s) in the overlying epithelium, and
3) no source of carcinoma in the adjacent structures. The
average age of patients with carcinoma in a dentigerous cyst
is 58.8 years, with a range from 25 to 84 years.[11]
Multiple dentigerous cysts have also been reported in the
literature. They are reported in patients with syndromes
like basal cell nevus syndrome,[2] mucopolysaccharidosis
type VI (Maroteaux-Lamy syndrome) and cleidocranial
dysplasia. [2,7] Bilateral mandibular dentigerous cysts
have been reported after prolonged concurrent use of
cyclosporine and calcium channel blockers.[2]
CONCLUSION
Dentigerous cysts are one of the most common
developmental odontogenic cysts, which are usually
detected in routine radiographic examination. Developing
dentigerous cysts are difficult to distinguish from normal
follicle. Pericoronal radiolucencies greater than 4 mm
are to be considered cystic, until proven otherwise.[9]
Diagnosis requires an accurate radiographic image to
be further confirmed by a histological examination. The
radiographic image should identify the enhancement of the
pericoronal space just around the crown of the impacted
tooth and will complement the histological examination
of the odontogenic cyst. [2] Daley and Wysocki have
recommended the following guidelines for the diagnosis
of a dentigerous cyst: 1) a pericoronal radiolucency >4
mm in greatest width, 2) histologically, fibrous tissue lined
Journal of Indian Academy of Oral Medicine and Radiology / October - December 2008 / Volume 20 / Issue 4
by nonkeratinized stratified squamous epithelium, and 3)
a surgically demonstrable cystic space between the enamel
and the overlying tissue. Of these, the third is the most
critical, but all the three must be satisfied.[6]
ACKNOWLEDGMENTS
Dr. T. K. Ramamurthy, Reader, Dr. T. A. Deepak, Senior Lecturer,
Department of Oral Medicine and Radiology, and my colleagues.
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Source of Support: Nil, Conflict of Interest: Nil
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