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Odontogenic Keratocyst: A Case Report In A Five-Year-Old Saudi Boy
Ahmed A. Zahrani, BDS, MSc, DFM
College of Dentistry, King Saud University, P.O. Box 60169, Riyadh 11545, Saudi Arabia.
An odontogenic keratocyst possesses tumor-like characteristics
because of its clinical behavior. The aggressive growth, high recurrence rate
and seeding of the cyst into soft tissue exemplify this behavior, ft has been
reported that the age distribution for keratocysts has a biomodal
characteristic, with the first and greater peak in the second and third decades
and a second lower peak in the fifth decade. Incidence of occurrence before age
ten is low. In this report, odontogenic keratocyst in a five-year-old Saudi boy
is presented and the management of the case is briefly discussed.
Ever since Philipsen1 described the odontogenic keratocyst in 1956,
the lesion has continued to raise a considerable clinical interest because of
its unusual growth pattern and tendency to recur after surgical removal.2-5 In view of this behavior, a precise
pre-operative diagnosis of the lesion is necessary to assure a proper surgical
approach and successful eradication.6-8
Odontogenic keratocysts occur over a wide age range with a
pronounced peak incidence in the second and third decades.-3,7,9-13 It is seen more frequently in males rather
than in females.7,11 The cystic lesion expands in a unicentric
ballooning pattern and enlarges predominantly in an anterior-posterior
direction. Radiographically, it appears as unilocular or multilocular
radiolucencies with well-defined sclerotic margin. The growth of odontogenic
keratocyst may occur rapidly in young children usually preventing teeth
eruption and mimicking often a dentigerous cyst.8
The purpose of this article is to present the clinical and
radiographical features of odontogenic keratocyst in a five-year-old patient. A
brief discussion concerning the management of this case is also included.
Case Report
A five-year-old boy reported in January 1992 to the Orai
Surgery Clinic at the College of Dentistry, King
Saud University
in Riyadh with
a chief complaint of swelling on the left side of the chin that caused a
considerable asymmetry of the face [Fig. 1 ]. As narrated by his father, the
swelling was noted six weeks previously while its size gradually, but
progressively, increased. It was painless and caused no alteration in sensation
over the mental nerve distribution. The labio-buccal sulcus was obliterated by
a bony swelling and fluctuation was elicited in its center [Fig. 2], Bony
expansion extended lingually and the overlying mucosa showed normal texture.
The adjacent teeth were all mobile and were assigned grade II to grade III
mobility. Radiographically, a well-circumscribed radiolucency extended from the
left second deciduous molar to the right deciduous canine involving almost the
whole height of the mandible [Fig. 3] and extended to a greater extent
labiobucally [Fig. 4]. The erupting permanent teeth were displaced from their
path of eruption and pathological resorption of the related deciduous teeth was
evident radiographically. Body scanning and physical examination ruled out the
existence of a basal-cell nevus syndrome. The cyst content was aspirated and
was of thick pus-like consistency. Electrophoresis of the cystic fluid
demonstrated low soluble protein content (< 3.75 g./lOO ml). The tentative
diagnosis of odontogenic keratocyst was made and confirmed afterwards by
histological examination of the surgical specimen. The cross section showed
parakeratinized stratified squamous epithelium with uniform thickness in most
areas, and with a prominent palsading basal layer, which was consistent with
the pre-operative diagnosis of odontogenic keratocyst [Fig. 5].
The case was managed conservatively by decompression of
the cystic cavity, followed six months later by enucleation and primary
closure, as a second stage procedure, in order to complete the treatment. The
decision of the second stage operation was made upon the deposition of bone
around the cystic wall and the steady diminishing in cystic cavity size. Eleven
months later, the osseous defect was completely filled in with normal bone
[Fig. 6].
Previous reports7-13 indicated that the incidence of odontogenic
keratocyst covers wide age ranges of seven to eighty-three years (Table 1). The
age of the patient was less than what had been reported previously. Rapid
growth in this case might be explained by the aggressive nature of this lesion
as reported earlier, particularly in young children.8
Various modes of therapy have been advocated for the
treatment of keratocyst and these include marsupialization, enucleation and
primary closure, enucleation with excision of the overlying mucosa and packing,
decompression with secondary enucleation, treatment of the whole cyst cavity
with Carnoy's solution prior to enucleation and radical surgery. In 1970,
Browne7 found no significant differences in recurrence
among these modalities but a recurrence rate of 0.0% with a mean follow-up of
7.4 years in a group of patients treated with Carnoy's solution as cauterizing
fixative agent,14 was reported.
Forssell and his coworkers15 found that the surgical difficulties in
removing the thin friable capsule and the existence of bony perforation that is
often associated with soft tissue adherence are the likely possible factors
responsible for recurrence. Nevertheless, their observation regarding the high
occurrence of recurring keratocyst in younger patients was not confirmed by
Vedtofte and Praetorius.10
Marsupialization is not the accepted treatment for
odontogenic keratocyst because pathological epithelium is left in situ. This
modality, however, remains the treatment of choice for large unilocular cyst
with extremely thin lining, provided it is followed by a second stage operation
consisting of enucleation with primary closure or packing.4 The simple conservative treatment employed in
this case was deliberate and regarded as the first stage of management. It was
instituted primarily to prevent the likely pathological fracture of the jaw
consequent to enucleation and to permit eruption of teeth involved in the
cystic process. The second stage procedure was undertaken when the cystic
cavity was eventually obliterated by the filling of bone, permitting teeth to
move up to where it could be aligned easily by orthodontic appliance.
A long post-operative follow-up over the years may
demonstrate the recurrence to be less dramatic. Therefore, an observation at
regular interval for a period of at least ten years is found to be very
necessary to make a diagnosis of recurrence without delay.7,15
- Philipsen
HP. Om keratocyster (kolesteatom, i kaeberne. Tandlaegebladet 1956;60:963.
- Pindborg JJ,
Hansen J. Studies
on odontogenic cyst epithelium. Acta Pathol Microbiol Sand
1963;58:283.
-
Toller PA. Origin and growth of cysts of the jaws. Ann R Coll Surg
Engl 1967;40:316-36.
-
Fickling BW. Cyst of the jaw, a long term survey of type and
treatment. Proc Roy Soc Med 1965;58:847-54.
-
Bramley PA, Browne RM. Recurring odontogenic cysts. Br J Oral Surg
1967,5:106-16.
-
Toller PA.
Newer concepts of odontogenic cysts. Int J Oral Surg 1972;1:3-16.
-
Browne RM. The odontogenic
keratocyst, clinical aspect. Br Dent j 1970;3:225-31.
-
Partridge
M, Towers JF, The primordial cyst (odontogenic keratocyst): Its tumor-like
characteristics and behavior. Br J Oral Maxillofac Surg 1987;25:271-79.
-
Payne TF.
An analysis of the clinical and histopathologic parameters of the odontogenic
keratocyst. J Oral Surg 1972;33(4):538-46.
-
Vedtofte
P, Praetorius F. Recurrence of the odontogenic keratocyst in relation to
clinical and histological features. A 20-year follow-up study of 72 patients.
Int J Oral Surg 1979;8:412-20.
-
Radden BG,
Reade PC. Odontogenic keratocysts. J Pathol 1973;5:325.
-
Angelopoulous
EK, Nicolatou O. Odontogenic keratocysts: clinicopathologic study of 87 cases.
J Oral Maxrllofac Surg 1990;48:593-99.
-
Nartey NO,
Saini T. Odontogenic keratocyst radiographic features. Saudi Dent J 1990;2:15-20.
-
Voorsmit
RACA. The incredible keratocyst: A new approach to treatment. Deutsch
Zahnaerztl Z 1985;40:641-44.
-
Forssell
K, Sorvari TE, Oksala E. An analysis of the recurrence of odontogenic
keratocyst ijaws. Proc Finn Dent Soc 1974;70:121-34.
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