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Case Report

Paradental cyst of the first molar: Report of


a rare case with bilateral presentation and
review of the literature

Borgonovo AE, Reo P, Grossi GB, Maiorana C


Abstract Department of Oral Surgery, University of Milan, Fondazione
IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Milan, Italy
The paradental cyst is a lesion classified and recognized by
World Health Organization quite recently, which is related Correspondence:
to an inflammatory process, especially pericoronitis, involving Dr. Pietro Reo, Via Wildt 14, 20131 Milan, Italy.
a tooth in eruption. The aim of this article is to report a rare E‑mail: pietroreo@hotmail.com
bilateral case of paradental cyst. An 8‑year‑old boy presented
to the Oral Surgery Department, Dental Clinic, Istituto di
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Ricerca e Cura a Carattere Scientifico (IRCCS) Fondazione
Quick Response Code: Website:
Ospedale Maggiore Policlinico, University of Milan, with
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the complaint of swelling over the buccal gingiva of his
DOI:
unerupted lower left first molar. Radiographs revealed a
10.4103/0970-4388.108940
radiolucency involving the bifurcation and root area of
PMID:
teeth 36 and 46. The cysts were enucleated, maintaining
***
the affected teeth in site; microscopic evaluation revealed a
chronically inflamed cyst lined by a non‑keratinized stratified
squamous epithelium; the histopathology associated with
macroscopic and radiographic examinations permitted the
According to the World Health Organization (WHO)
definitive diagnosis of a paradental cyst on the mandibular histological typing of odontogenic tumors, the
left and right first molars. The most recent literature shows paradental cyst is defined as “a cyst occurring near
the rarity of the paradental cyst occurring with bilateral to the cervical margin of the lateral aspect of a root
localization. Because the paradental cyst can present variable as a consequence of an inflammatory process in a
clinical and radiographic signs, it is mandatory to correlate periodontal pocket. A distinctive form of the paradental
all clinical, radiographic, and histological data to obtain a cyst occurs on the buccal and distal aspects of erupted
definitive diagnosis. mandibular molars, most commonly the third molars,
where there is an associated history of pericoronitis.”[3]
Key words
Bilateral cyst, mandibular disease, oral surgery, paradental cyst The etiology of these cysts is still debated, but it
is believed that they originate from the reduced
epithelium of enamel[2,4] or from the inflammatory
Introduction proliferation of epithelial rests of Malassez [5,6]
that come from the superficial mucosa of a tooth in
The first clinical and histological description of a eruption (pericoronitis).[3] They represent beyond 5%
paradental cyst was reported by Main[1] in 1970. The of all odontogenic cysts.[2,7]
condition was initially described as an inflammatory
collateral cyst, but the diagnosis has caused many The major clinical feature of the paradental cyst is
controversies in the literature. The current nomenclature the presence of a recurring inflammatory periodontal
was suggested by Craig[2] in 1976. process, usually a pericoronitis. Apart from acute

JOURNAL OF INDIAN SOCIETY OF PEDODONTICS AND PREVENTIVE DENTISTRY | Oct - Dec 2012 | Issue 4 | Vol 30 | 343
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Borgonovo, et al.: Bilateral paradental cyst of the first molar

episodes, this cyst presents only a few signs and mild and then enucleated trough the access previously
symptoms, including discomfort, tenderness, moderate created [Figure 5]. Having done an irrigation with
pain, and in some cases, suppuration through the sterile saline, the suture was carried out with silk 4/0.
periodontal sulcus.[8‑10]
Histologically, the cyst capsule was lined by a
The aim of this work is to present a rare case of bilateral proliferating, non‑keratinized, stratified squamous
paradental cyst in a young patient affecting the buccal epithelium, showing as arcading. The cystic wall
aspect of lower first molars, discussing diagnosis, consisted of a dense, mature fibrous connective tissue,
treatment, and radiographic findings of the case. with an intense chronic inflammatory reaction mainly
near the epithelium [Figure 6].
Case Report
An 8‑year‑old boy was referred to Oral Surgery
Department, Dental Clinic, IRCCS Fondazione
Ospedale Maggiore Policlinico, University of Milan,
Italy, with the complaint of swelling over the buccal
gingiva of his unerupted lower left first molar. Clinically,
there was mild edema in the overlaying mucosa distal
to the second deciduous molar, showing a bluish
color [Figure 1]. The evaluation of the panoramic
radiography [Figure 2] disclosed a well‑defined
semilunar‑shaped radiolucency, demarcated by a fine
radiopaque line, on the buccal aspect of the unerupted Figure 1: Intraoral pre‑operative view – left side
lower left first molar [Figure 3]. Observing closely the
radiograph, a second lesion was evident on the buccal
aspect of the partially erupted right lower first molar,
but less defined than the previous.

Computed tomography showed and confirmed


the presence of bilateral lesions to the lower first
molars [Figures 4]; in both cases, the margins of
radiolucent lesions were delimited by a thin layer of
denser bone on the buccal aspects, revealing the presence
of the cysts and their relations to both first and right
lower first molars; the cysts were extended from the
cement–enamel junction to the lower root margins, Figure 2: Panoramic radiograph shows a well‑defined semilunar‑shaped
radiolucency, demarcated by a fine radiopaque line on the buccal
measuring about 10 mm in the largest extension. aspect of the left and right lower first molars

Electric pulp test for left lower first molar, partially


erupted, was positive.
Clinical, radiographic, and anamnestic findings
suggested an initial diagnosis of paradental cyst.
We decided for a surgical removal of the cysts under
general anesthesia, planning to maintain the affected
teeth.
The surgical approach was a full‑thickness trapezoidal
flap, with gingival crevicular incision and vertical
releasing incisions; buccal ostectomy was done, care
was taken to preserve a sufficient band of cortical bone
in the coronal aspect. The cysts have been exposed Figure 3: Intraoral pre‑operative view – right side

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Borgonovo, et al.: Bilateral paradental cyst of the first molar

The histopathology associated with macroscopic and about 1 year after surgery, evidence the complete
radiographic examinations permitted the definitive regression of the lesion and the correct eruption
diagnosis of a paradental cyst on the mandibular left of the mandibular permanent left and right first
and right first molars. molars [Figures 7‑8].

Follow‑up is still ongoing, but the panoramic Discussion


radiography and clinical examinations, performed
An inflammatory etiopathogenic nature of paradental
cysts has been widely discussed in the literature. Initially,

Figures 5: Intraoperative view of the enucleation of the two cysts

Figure 6: Histopathological analysis of the two cysts

Figures 4: Computed tomography shows that the margins of Figure 7: Panoramic radiograph  1  year after surgery, indicating a
radiolucent lesion were delimited by a thin layer of denser bone on complete regression of the two lesions and the correct eruption of the
the buccal aspect mandibular left and right first molars

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Borgonovo, et al.: Bilateral paradental cyst of the first molar

The paradental cyst is localized exclusively in the


mandibular region, almost always on the distal or
vestibular side of a completely or partially erupted
molar, but always vital. Although the mesial surface
could be involved very rarely,[5,9] the lingual aspect
is never interested. More than 60% of the cases of
paradental cysts are associated with the lower third
molars.[9]

Since the radiological features are different according to


the tooth involved,[9] we can distinguish the paradental
cysts developed on the first and second inferior molars,
also called as “juvenile paradental cysts,”[1] from those
Figures 8: Intraoral view 1 year after surgery, showing the correct involving the inferior third molar.
eruption of mandibular left and right first molars
The medium age of the patients with paradental cyst
all reported cysts involved lower third molars with localized at the lower first molar is 8‑9 years, whereas
inflammatory processes[11] and when the lower first cysts localized to the second molar appear between
permanent molars of children aged between 6 years and 13 years and 20 years of age. Bilateral localizations are
8 years were involved, these cysts were called mandibular marked in 23.6% of the cases.[9]
infected buccal cysts.[12] This type of cystic lesion was
considered a distinct clinical entity by some authors,[4,13] The clinical symptoms and signs are those of the
but this concept was never fully accepted.[11] periodontitis and are common to both localizations,
while the onset of a vestibular swelling seems to be
The paradental cyst is considered a rare lesion; it associated exclusively to the paradental cysts that
was included in the WHO histological typing of involved the first molar.[9]
odontogenic tumors for the first time in 1992, although
it has been described in several clinicopathological Since the lesion is localized on vestibular aspect of the
studies in specialized journals since 1970.[1,2,5,6] The roots, the involved molar is usually tilted so that the
relatively recent characterization of this cyst can be a root apices are adjacent to the lingual cortex with the
contributing factor to its non‑recognition; on the other crown showing buccal tipping.[17]
hand, it has been speculated that this lesion has been
underdiagnosed. Lindh and Larsson[14] believe that the The nearly exclusive involvement of the vestibular
paradental cyst has been misdiagnosed as a dentigerous surface would be explained by the fact that the
cyst, lateral radicular cyst, or merely as pericoronitis or mesio‑buccal cuspids are the first to perforate the oral
some other entity related to inflammatory conditions mucosa during the eruption, and therefore, the first
of the dental follicle. Another fact that could result to be exposed to the oral ambient, as described by
in the underdiagnosis of paradental cysts is that Stoneman et al.[10]
histopathological analysis of extirpated follicular sacs
is rarely done. It has been stated that the radiographic image of the
paradental cyst involved the first or second molar, and
The prevalence of paradental cysts is low compared is always characterized by a well‑defined radiolucency
to other cysts, representing 3‑5% of all odontogenic associated with the roots on the buccal aspect.[2,6]
cysts.[4,5,12,15] In the mandible, this lesion was detected
in only 26 (0.9%) of the 2700 cyst cases studied by The medium age of the patients with paradental cysts
Magnusson and Borrman,[16] who ascribed the low localized at the lower third molar is approximately
prevalence to several possible misdiagnoses. Specifically 25‑30  years, with a distal or disto‑vestibular localization
in regard to the lower third molars, this can be to the affected tooth; bilateral localizations are marked
considered the second most frequent cyst, representing just in 4.1% of the cases.[9] In these cases of paradental
up to 25% of the cystic lesions associated with these cyst, usually a history of recurrent pericoronitis
teeth, although they represented only 1.6% of the cystic is reported and there is often the presence of a
lesions analyzed by Colgan et al.[4] communication between the periodontal pocket and

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Borgonovo, et al.: Bilateral paradental cyst of the first molar

the cyst. The cortical expansion of the bone is not pulp test is a diagnostic criterion for paradental cyst.
so frequent like in the forms previously described, The diagnosis would be a lateral radicular cyst if the
and the majority of lesions do not exceed 15 mm of associated tooth is non‑vital.[21]
diameter.[18]
The initial diagnosis of paradental cyst was made
The etiology of paradental cysts is of an inflammatory considering the anamnestic, clinical, and radiological
nature, as shown by the histological findings of features. Only the enucleation of cyst without
odontogenic epithelium proliferation, presence of extraction of the adjacent tooth was done in both
an inflammatory infiltrate, and occasional hyaline the cysts. Most reports[5,11,20‑22] show that if the tooth
changes in blood vessel walls.[4,13,15‑17] However, there involved is the first or the second molar, the treatment
are controversies surrounding the origin of the lining of choice is enucleation of the cyst without the
epithelium. According to Souza et al.,[15] most cases of extraction of the tooth, whereas surgical removal of
paradental cysts stem from the proliferation of reduced the tooth and the paradental cyst has been considered
epithelium of the enamel organ, probably caused by the best case solution when the involved tooth is
inflammatory stimuli originating from the junction of a third molar. [8,9,21] Pompura et  al. [23] presented
the epithelium of the cystic capsule with the gingival 44 cases treated by enucleation without extraction.
epithelium. Colgan et al.[4] and Lim and Peck[17] also Packota et  al.[11] successfully treated five cases of
believe that this cyst arises from reduced epithelium of paradental cyst involving the mandibular first molar
the enamel organ. The epithelial remnants of Malassez with enucleation of the cyst without extraction. In Wolf
seem to be the most unlikely origin, although they may and Hietanen’s report,[20] of all the cases of mandibular
unreasonably explain cysts located near the roots.[15] infected buccal cyst (paradental) associated with the
first molar (three cases) and the second molar (three
In this article, the authors present a case of bilateral cases), four were treated without extraction. In Vedtofte
paradental cyst involving both the left and right and Praetorius’s[21] series involving the mandibular
mandibular first molar. In both cases, the radiographic first and second molars, 11 of the 13 cases treated
image of these lesions revealed a well‑defined with preservation of the involved tooth had successful
radiolucency associated with the roots on their buccal outcome. In all cases, recurrence is rare, provided that
aspect. the lesion has been completely removed.[6,21,23‑25]

The differential diagnosis included the radicular cyst, The histopathological features of the paradental cyst
odontogenic keratocyst, lateral periodontal cyst, are identical to the radicular cyst and to those of
gingival cyst, dental follicles, and the dentigerous cyst. other inflammatory odontogenic cysts; microscopic
examination shows a fibrous connective tissue capsule
The most recent literature shows the rarity of the invaded by a lymphocytic inflammatory infiltrate, lined
paradental cyst occurring with bilateral localization; by a hyperplastic, non‑keratinized, stratified squamous
some authors[17] report only seven cases of bilateral epithelium; in the case that we described, both the cysts
involvement, including lesions occurring on first, capsule were lined by a proliferating, non‑keratinized,
second, and third molars; a more detailed analysis on stratified squamous epithelium, showing as arcading.
342 cases carried out by Philipsen et al.[9] reveals only The cystic wall consisted of a dense, mature fibrous
23.6% of bilateral occurrence for paradental cysts on connective tissue, with an intense chronic inflammatory
first and second mandibular molars and 4.1% for those reaction mainly near the epithelium, supporting a
involving third molars. Considering the low prevalence definitive diagnosis of a paradental cyst.
of the paradental cyst (3‑5% of all odontogenic cysts),
we can assert that the bilateral occurrence is very rare. The paradental cyst can present variable clinical and
radiographic signs,[2] in addition to being confounded
In our case, the second lesion on the right side with the radicular cyst at the microscopic level; for
was noted on radiographs (and confirmed only by these reasons, it is mandatory to correlate all clinical,
computed tomography); for this reason, it is generally radiographic, and histological data to obtain a definitive
recommended that the contra‑lateral tooth should be diagnosis. Surgical findings, such as bony cavitation,
carefully evaluated for a second lesion. cystic content, and location of lesion adherence,
can give some important clues. Enucleation of the
Most studies[5,6,10,19,20] report that a positive electric lesion with the maintenance of the associated tooth

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Borgonovo, et al.: Bilateral paradental cyst of the first molar

can be indicated when the first or second molars are Surg 1990;48:258‑63.
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Clinicopathologic features of 54 cases of paradental cyst.
Quintessence Int 2001;32:737‑41.
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