Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx

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Journal of Oral and Maxillofacial Surgery, Medicine, and

Pathology
journal homepage: www.elsevier.com/locate/jomsmp

Case report

A dentigerous cyst associated with a supernumerary tooth (fourth molar) in

the mandibular ramus: A case report
⁎
Kenji Haraa, Shuhei Tsuchiyaa, , Sumitaka Hagiwarac, Masahito Fujiob, Akira Sayob,
Hideharu Hibib
a
Department of Oral and Maxillofacial Surgery, Nagoya University Hospital, 65 Tsurumai-cho, Showa-ku, Nagoya 466-8550, Japan
b
Department of Oral and Maxillofacial Surgery, Nagoya University Graduate School of Medicine, 65 Tsurumai-cho, Showa-ku, Nagoya 466-8550, Japan
c
Department of Head and Neck Surgery, Aichi Cancer Center, 1-1 Kanokoden, Chikusa-ku, Nagoya 464-8681, Japan

A R T I C LE I N FO A B S T R A C T

Keywords: Dentigerous cysts (DC) are the second most common odontogenic cysts of the jaw and are usually associated
Dentigerous cyst (DC) with the crowns of impacted or unerupted teeth. In particular, DCs tend to occur around unerupted mandibular
Mandibular third molars and maxillary mesiodens, which are supernumerary teeth. Such cases account for 5% of all DCs.
Supernumerary tooth However, DCs related to the supernumerary (fourth) molar distal to the mandibular third molar are uncommon,
Fourth molar
occurring in 0.04–0.175% of investigated populations. This article reports the case of a 43-year-old woman with
Impacted tooth
a DC associated with a mandibular supernumerary (fourth) molar.

1. Introduction 2. Case report

A dentigerous cyst (DC) is a cystic lesion enclosing the crown of an A 43-year-old Japanese woman attended our clinic for a standard
erupted tooth [1]. DCs are the second most common type of odonto- dental examination. Intraoral examination revealed that the third molar
genic cyst after radicular cysts [2] and are typically asymptomatic. on the right mandible had completely erupted into the oral cavity. A
However, without treatment, DCs often develop into large, destructive, panoramic radiograph showed a fourth molar on the right mandibular
expansile lesions [1]. The pathogenesis of DCs is generally considered ramus, with a well-defined radiolucent area encompassing the crown
to be developmental in nature, although an inflammatory etiology has (Fig. 1). The patient reported no pain or discomfort around the lower
been suggested in some cases [3,4]. Among all jaw cysts, the prevalence fourth molar, and no anatomic changes were observed around the root
of DCs is 16.6%, with the most common site being the mandibular third of the third molar (Fig. 1). A computed tomography (CT) scan was
molar followed by the maxillary mesiodens, which are supernumerary performed to determine the relationship of the fourth molar to the
teeth [5], that is, in addition to the normal complement of teeth [6]. anatomical structure, as well as the extent of the radiolucent area
Supernumerary teeth have been reported in both the primary and around the tooth. The CT examination revealed that the fourth molar
permanent dentition. The prevalence of supernumerary teeth in the was positioned at the anterior border of the mandibular ramus
permanent dentition is estimated to range from 0.1% to 3.8% [6,7]. (Fig. 2A). An orbicular space enveloping the crown of the tooth was also
Supernumerary teeth are most commonly located in the premaxillary observed, suggesting that the cyst extended to the lingual cortical bone
region, and 92.8% occur in the central incisor [8,9]. In the mandible, of the mandible (Fig. 2B). The patient was referred to a specialist at the
especially among mandibular molars, supernumerary teeth are rare. Central Block of Radiology, Nagoya University Hospital. The medical
Although there are previous reports of a fourth molar [10–16], reports radiologist reported that CT images revealed a supernumerary tooth
of a fourth molar associated with a DC are limited [10,17–20]. located horizontally on the dorsal side on the right mandibular third
Here, we discuss a rare impacted supernumerary fourth molar in the molar with an orbicular radiolucent area measuring 12 × 12 mm as-
mandibular ramus. The DC was not associated with any underlying sociated with the tooth crown. The tooth was observed to be close to the
disease. We also discuss how the case was managed and explained to mandibular canal, but they were not in contact. On the basis of these
the patient. findings, the patient was given a clinical and radiological diagnosis of

⁎
Corresponding author at: Department of Oral and Maxillofacial Surgery, Nagoya University Graduate School of Medicine, 65 Tsurumai-cho, Showa-ku, Nagoya,
Aichi 466-8550, Japan.
E-mail address: t-shuhei@med.nagoya-u.ac.jp (S. Tsuchiya).

https://doi.org/10.1016/j.ajoms.2018.08.005
Received 20 February 2018; Received in revised form 18 June 2018; Accepted 13 August 2018
2212-5558/ © 2018 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd All rights reserved.

Please cite this article as: Hara, K., Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology (2018),
https://doi.org/10.1016/j.ajoms.2018.08.005
K. Hara et al. Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx

Fig. 1. Preoperative orthopantomograph showing a cystic lesion in the mandibular fourth molar. (A) The impacted fourth molar in the mandibular ramus. (B) A
radiolucent region around the crown of the tooth. (C) No root resorption in the third molar.

Fig. 2. Preoperative CT images. (A) Coronal section showing the dentigerous cyst encompassing the crown of the fourth molar. (B) Axial section showing the cyst
extending to the lingual cortical bone of the mandible.

DC. A treatment plan was prepared that comprised cyst enucleation Immunohistochemical staining with PCNA and Ki-67 showed that
with extraction of the impacted supernumerary tooth, followed by PCNA positive cells were mainly observed in the basal layer and prickle
histological examination of the enucleated cyst. cell layer and that Ki-67-labeled nuclei were concentrated in the basal
The patient was anesthetized using local anesthesia combined with layer. The percentages of PCNA-positive cells and Ki-67-labeled nuclei
intravenous sedation with midazolam. The oral cavity was accessed were 14.7% and 5.3%, respectively. Thus, the final diagnosis of DC was
posterior to the third molar through incisions in the buccal gingiva and made on the basis of the histological examination, clinical course, and
the gingival sulcus of the third molar. Periosteal dissection was per- operative findings (Fig. 4).
formed to expose the buccal side of the mandible. The approximate At the 1 year follow-up appointment, our patient reported no pain
locations of the cyst and the crown of the supernumerary tooth were or discomfort around the right mandibular ramus. A radiographic ex-
determined from the panoramic radiograph and CT scans, and the cyst amination revealed that radiopacity had increased around the region
site was accessed using a dental bur. After a cut was made between the where the supernumerary tooth and associated DC had been located
crown and the root, the tooth was elevated out of its bony socket using (Fig. 5).
an elevator, and the cyst was then gently enucleated (Fig. 3). The cystic
cavity was curetted and irrigated using saline, and the wound was then
sutured using 3-0 silk sutures. The enucleated soft tissue was sent to the 3. Discussion
Department of Pathology and Laboratory Medicine, Nagoya University
Hospital. At the 1 week follow-up appointment, the wound was clear Of odontogenic cysts, DCs are the second most common type after
and no swelling or pain was observed. radicular cysts [2]. DCs occur most often around the mandibular third
In the histopathological examination of the excised tissue, hema- molar, followed by supernumerary teeth [5,10,21]. It has been reported
toxylin and eosin staining showed that the lesion comprised a fibrous that DCs occasionally occur around odontomas [22] and fused teeth
cyst wall covered with non-keratinized squamous epithelium and the [23,24]. Supernumerary teeth are most frequently observed in the
presence of mucus-containing cells in the epithelium. maxilla, and their prevalence varies between 0.1% and 3.8% in the
permanent dentition [6]. One previous report stated that mesiodens are

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Fig. 3. Macro photographs. (A) No enamel hypoplasia was observed on the removed supernumerary tooth. (B) Enucleated cyst encompassing the crown of the fourth
molar during surgery.

the most common supernumerary teeth followed by premolars, lateral fourth molar have been reported in the English and Japanese literature
incisors, and fourth molars [25]. In the literature, several cases of a [10,17–20] (Table 1).
mandibular fourth molar have been reported [10–16,25]. Nevertheless, Odontogenic cysts are usually accompanied by displacement of the
fourth molars in the mandible are rare and occur in only 0.04–0.175% teeth associated with the lesion. In the present case, the cyst was not
of the investigated population [16,25]. well developed, and both radiographic and intraoperative findings
DCs around supernumerary teeth account for about 5% of all DCs showed that it had displaced the fourth molar only slightly within the
[21]. Previous studies have revealed that the mean age of DC patients is mandibular ramus. At the time of surgery, the follicle was small and did
25.7–30 years, with 69.6–82% of patients being within the first four not contain sufficient fluid to warrant tentative aspiration. Because
decades of life. Moreover, there is a male-to-female ratio of odontogenic keratocysts and ameloblastomas may present similarly in
1.75–2.80:1, with 84.8–90% of cysts originating from maxillary me- radiographic images [26–28], fluid aspiration of the cyst contents, as
siodens [5,21]. well as histological examination, must be performed to exclude these
Given the low prevalence of mandibular fourth molars, the occur- and other possible lesions.
rence of a DC associated with a mandibular fourth molar is quite rare. In the histopathological examination of the excited specimen, he-
Besides the present case, DCs associated solely with a mandibular matoxylin and eosin staining suggested a cystic lesion. In addition, the

Fig. 4. Histopathology of hematoxylin and eosin staining and immunohistochemical staining with PCNA and Ki-67 of the cyst tissue. (A) Hematoxylin and eosin
staining showed that cyst walls were composed of fibrous tissue and lined with squamous epithelium consisting of several layers, as well as the existence of mucus-
containing cells in the epithelium (arrow), (B) PCNA-positive cells were mainly observed in the basal layer and the prickle cell layer. (C) Ki-67-labeled nuclei were
concentrated in the basal layer.

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Fig. 5. Orthopantomograph at the 1 year follow-up appointment showing increased radiopacity within the region where the supernumerary fourth molar and
associated DC had been enucleated.

Table 1
Published case reports of DCs associated with a mandibular fourth molar.
Author Year Age Gender Position Treatment

Kanematsu et al. [17] 1982 28 Male Left Enucleation of cyst, third molar and supernumerary tooth removal
Uesugi et al. [18] 2008 45 Female Left Enucleation of cyst and supernumerary tooth removal
McCrea et al. [10] 2009 60 Female Right Enucleation of cyst, third molar and supernumerary tooth removal
Gulses et al. [19] 2012 27 Male Right Surgical removal
Sanghera et al. [20] 2013 49 Female Right Surgical removal from exraoral approach; preauricular

percentage of PCNA-positive cells was high, compared with a previous cyst was not large. For these reasons, the patient was not aware of the
report of 5.3% in the epithelium of DCs [29]. PCNA is an important lesion until she underwent radiographic examination.
protein in DNA synthesis and repair, and it is present in cells from the DCs are often similar to ameloblastomas and keratocystic odonto-
G1 phase to early S phase [30]. Excess PCNA has been reported in S genic tumors in terms of their radiographic features. Thus, DCs must be
phase cells [31], and it has been suggested that PCNA is also present in definitively diagnosed by histopathological examination. In patients
cells that have completed the cell cycle. In the current case, the per- who do not receive regular dental healthcare, these cysts are likely to
centage of Ki-67-labeled nuclei was 5.33%, which is consistent with a grow as they are often asymptomatic. In such cases, invasive treatment
previous report [32]. Because Ki-67 is expressed in proliferating cells may be necessary as soon as the lesion is diagnosed. Early recognition
and during DNA synthesis and immediately disappears after mitosis, it of lesions depends on where the DC has occurred, so periodic dental
is very useful as a cell proliferation maker. These findings suggest that examination and radiography are important, particularly for patients
cells in the epithelium are less likely to proliferate early as neoplastic with jaw abnormalities.
lesions. At the 1 year follow-up appointment, the patient reported no pain or
DCs originate because of alteration of the reduced enamel epithe- discomfort around the right mandibular ramus, and radiographic ex-
lium after the crown of the tooth has completely formed, resulting in amination revealed that radiopacity had increased within the region of
accumulation of fluid between the reduced enamel epithelium and interest. Hence, recurrence of the lesion was not suspected at that time.
tooth crown. If the stellate reticulum degenerates before the tooth
crown has completely formed, the result is a cyst associated with a
tooth exhibiting enamel hypoplasia [33]. In one report, 43 of 86 teeth 4. Conclusion
with DCs showed enamel hypoplasia on the occlusal surfaces of their
incisal edges [34]. However, in the present case, there was no enamel Here, we report a case of a DC associated with a supernumerary
hypoplasia on the removed supernumerary tooth, indicating that the (fourth) molar in the mandibular ramus of a 43-year-old woman.
cyst had formed as a result of fluid accumulation in the reduced enamel Occurrence of a DC in this region is quite rare. In such rare cases, di-
epithelium after complete crown formation. agnosis at an early stage with properly planned treatment is important
Because most supernumerary teeth are located in the premaxilla, it for preventing complications.
follows that DCs are most likely to occur in cases of maxillary mesio-
dens. As most DCs are asymptomatic, they are often diagnosed during
radiographic examination as a part of periodic dental healthcare or Ethical approval
because of swelling related to the growing cyst [35,36]. In the present
case, the DC occurred in the mandibular ramus, which is rare, and the The patient provided informed consent prior to submission of this
paper.

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