Unusual Imaging Features of Dentigerous Cyst: a Case Report

Case Report Unusual Imaging Features of Dentigerous Cyst: A Case Report

Carla Patrícia Martinelli-Kläy 1,2,*, Celso Ricardo Martinelli 2, Celso Martinelli 2, Henrique Roberto Macedo 2 and Tommaso Lombardi 1

1 Laboratory of Oral & Maxillofacial Pathology, Oral Medicine and Oral and Maxillofacial Pathology Unit, Division of Oral Maxillofacial Surgery, Department of Surgery, Geneva University Hospitals, University of Geneva, 1211 Geneva, Switzerland 2 Centre for Diagnosis and Treatment of Oral Diseases, Ribeirão Preto 14025-250, Brazil * Correspondence: [email protected]; Tel.: +41-22-379-4034

Received: 26 March 2019; Accepted: 5 July 2019; Published: 1 August 2019

Abstract: Dentigerous cysts (DC) are cystic lesions radiographically represented by a well-defined unilocular radiolucent area involving an impacted tooth crown. We present an unusual radiographic feature of dentigerous cyst related to the impacted mandibular right second molar, in a 16-year-old patient, which suggested an ameloblastoma or odontogenic keratocyst (OKC) because of its multilocular appearance seen on the panoramic radiography. A multi-slice computed tomography (MSCT), however, revealed a unilocular lesion without septations, with an attenuation coefficient from 3.9 to 22.9 HU suggesting a cystic lesion. Due to its extension, a marsupialization was performed together with the histopathological analysis of the fragment removed which suggested a dentigerous cyst. Nine months later, the lesion was reduced in size and then totally excised. The impacted mandibular right second molar was also extracted. Histopathological examination confirmed the diagnosis of a dentigerous cyst. One year later, the panoramic radiography showed a complete mandible bone healing. Large dentigerous cysts can sometimes suggest other more aggressive pathologies. Precise diagnosis is important to avoid mistakes since DC, OKC and ameloblastoma require different treatments. Histological examination is, therefore, essential to establish a definitive diagnosis. In our case, MSCT and the tissue attenuation coefficient analysis contributed to guide the diagnosis and management of the dentigerous cyst.

Keywords: dentigerous cyst; radiographic diﬀerential diagnosis; multislice computed tomography; hounsﬁeld unit analysis

1. Introduction A dentigerous cysts (DC) are defined as cystic lesions involving the crown of impacted teeth caused by fluid accumulation between the follicular epithelium and the crown of the tooth. It is considered the most common type of noninflammatory odontogenic cyst, and it occurs mainly in the lower third molar tooth of male patients with peak incidences in adolescents or young adults. Dentigerous cysts present a slow painless swelling and can cause the teeth displacement or teeth and bone resorption. Large cysts, however, may be associated with pain [1–4]. Histologically, they are represented by a cavity lined by the non-keratinizing thin epithelium without rete pegs. Their wall is usually fibrous and devoid of inflammatory cells [1]. Radiographically, DCs often present a unilocular radiolucent area around the crown of the impacted tooth surrounded by a well-defined sclerotic area [1,2,4,5]. Large cysts may cause cortical bone expansion. Moreover, DCs can rarely show a multilocular feature in the panoramic radiography. This is probably due to the cyst growth in areas of different bone densities [2]. In this case, the

Dent. J. 2019, 7, 76; doi:10.3390/dj7030076 www.mdpi.com/journal/dentistry Dent. J. 2019, 7, 76 2 of 7 diffDent.erential J. 2019 diagnosis, 7, x FOR PEER should REVIEW be made with other more aggressive lesions, such as ameloblastomas,2 of 7 odontogenic keratocysts (OKCs), other odontogenic tumours [1–3]. case,Computed the differential tomography diagnosis (CT), such should as Cone be made Beam with CT (CBCT)other more or multi-slice aggressive computed lesions, tomography such as ameloblastomas, odontogenic keratocysts (OKCs), other odontogenic tumours [1–3]. (MSCT), has an important application in the evaluation of head and neck lesions. It is a non-invasive Computed tomography (CT), such as Cone Beam CT (CBCT) or multi-slice computed technique that permits the lesion size and margins, the bone destruction and expansion patterns to be tomography (MSCT), has an important application in the evaluation of head and neck lesions. It is a precisely analysed [6,7]. In addition, MSCT provides an accurate measurement of the tissue attenuation non-invasive technique that permits the lesion size and margins, the bone destruction and expansion coepatternsfficient [to7– be10 ].precisely Each tissue analysed has [6,7 the]. abilityIn addition, to absorb MSCT a provides certain proportionan accurate ofmeasurement X-rays as theof the bone, fortissue example, attenuation which absorbscoefficient a lot[7– of10]. X-rays Each tissue while has the the air ability almost to none. absorb An a arbitrarycertain proportion scale named of X- the Hounsfield scale shows these attenuation coefficients: 1000 Hounsfield unit (HU) represents the air rays as the bone, for example, which absorbs a lot of X−-rays while the air almost none. An arbitrary attenuation,scale named 0 is the the Hounsfield water attenuation scale shows and these+1000 attenuation HU is the coefficients: bone attenuation −1000 Hounsfield [7,8]. unit (HU) representsThis paper the illustrates air attenuation, a case 0 ofis anthe unusualwater attenuation dentigerous and cyst +1000 initially HU is diagnosedthe bone attenuation as ameloblastoma [7,8]. due to itsThis multiloculated paper illustrates feature a case observed of an unusual in panoramic dentigerous radiography. cyst initially diagnosed as ameloblastoma due to its multiloculated feature observed in panoramic radiography. 2. Case Presentation 2. Case Presentation A 16-year-old male was referred to the Centre for Diagnosis and Treatment of Oral Diseases with a clinicalA diagnosis16-year-old of male ameloblastoma was referred havingto the Centre hemi-mandibulectomy for Diagnosis and asTreatment the suggested of Oral therapy. Diseases At admission,with a clinical no relevant diagnosis aspects of ameloblastoma within his medical having hemi history-mandibulectomy were observed. as Hethe broughtsuggested a panoramictherapy. radiographyAt admission, (Figure no relevant1) which aspects revealed within the his presence medical ofhistory a multilocular were observed. lesion He involving brought a the panoramic impacted mandibularradiography right (Figure second 1) molarwhich suggestingrevealed the mainly presence an of ameloblastoma a multilocular orlesion an OKC. involving It extended the impacted from the rightmandibular mandibular right notch second to the molar lower suggesting right first mainly premolar. an ameloblastoma A displacement or an of theOKC. mandibular It extended canal from and thethe lower right right mandibular third molar notch towards to the lower the mandibular right first pr notchemolar. was A alsodisplacement observed. of We the had mandibular access to canal another and the lower right third molar towards the mandibular notch was also observed. We had access to radiography taken at the age of thirteen, which presented a well-defined sclerotic area around the another radiography taken at the age of thirteen, which presented a well-defined sclerotic area crown of the mandibular right second molar suggesting a hyperplastic dental follicle or a dentigerous around the crown of the mandibular right second molar suggesting a hyperplastic dental follicle or cyst (Figure2). During those three years, the patient did not consult. Intraoral examination revealed a a dentigerous cyst (Figure 2). During those three years, the patient did not consult. Intraoral slightexamination painless swellingrevealed a of slight thebuccal painless and swelling lingual of corticalthe buccal extending and lingual from cor thetical lower extending right from first molarthe regionlower to right the mandibular first molar region branch to (notthe mandibular shown). branch (not shown).

FigureFigure 1. Panoramic 1. Panoramic radiography: radiography A well-deﬁned: A well multiloculated-defined multiloculated radiolucent lesion radiolucent involving lesionimpacted mandibularinvolving right impacted second mandibular molar. right second molar.

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Figure 2. Panoramic radiography made at the age of thirteen: A unilocular well-defined Figure 2. Panoramic radiography made at the age of thirteen: A unilocular well-defined radiolucent lesionradiolucent (measuring lesion around (measuring 4 mm) surrounding around 4 impactedmm) surrounding mandibular impacted right second mandibular molar. right Figure 2. Panoramic radiography made at the age of thirteen: A unilocular well-defined second molar. radiolucent lesion (measuring around 4 mm) surrounding impacted mandibular right With the aim of further analysis of the lesion, an MSCT (Figure3) was carried out, and unlike the second molar. panoramicWith radiography, the aim of further it showed analysis an of area the of lesion, hypoattenuation an MSCT (Figure without 3) was septations. carried out, The and Hounsfield unlike the panoramic radiography, it showed an area of hypoattenuation without septations. The unit (HU)With value the in aim the of lesion further varied analysis from of the 3.9 lesion, to 22.9 an HU, MSCT which (Figure suggested 3) was carried a lesion out, containing and unlike liquid thatHounsfield couldthe panoramic be compatible unit (HU) radiography, value with ain dentigerous it the showed lesion anvaried cyst area or from evenof hypoattenuation3.9 a unicystic to 22.9 HU, ameloblastoma. without which suggestedseptations From . a this Tlesionhetissue attenuationcontainingHounsfield coe liquidffi unitcient that (HU) analysis, could value be acompatibleinlesion the lesion such with varied as a an dentigerous ameloblastoma from 3.9 to cyst 22.9 or (solid HU,even which /amulticystic unicystic suggested ameloblastoma. type), a lesion or a cystic From this tissue attenuation coefficient analysis, a lesion such as an ameloblastoma (solid/multicystic lesioncontaining containing liquid a cheese-like that could be substance compatible as with found a dentigerous in odontogenic cyst or even keratocyst a unicystic could ameloblastoma. be disregarded. type), or a cystic lesion containing a cheese-like substance as found in odontogenic keratocyst could BecauseFrom of this the tissue lesion attenuation extension, coefficient marsupialization analysis, a waslesion performed such as an ameloblastoma together with (s theolid/multicystic histopathological be type)disregarded, or a cystic. Because lesion containingof the lesion a cheese extension,-like substance marsupialization as found wasin odontogenic performed keratocyst together withcould the analysis of the fragment removed, which suggested a dentigerous cyst. During this procedure, a clear histopathologicalbe disregarded. analysisBecause ofof the lesionfragment extension, removed, marsupialization which suggested was aperformed dentigerous together cyst. During with the this and pale fluid flowed out of the cavity. The lesion was irrigated weekly with chlorhexidine gluconate procedure,histopathological a clear analysis and pale of fluidthe fragment flowed removed, out of the which cavity. suggested The lesion a dentigerous was irrigated cyst. Duringweekly this with 0.12%chlorhexidineprocedure, oral solution. a gluconate clear During and 0.12% pale treatment, fluid oral solution flowed a candida out. During of( Candida the treatment, cavity.sp.) The a infection candida lesion was (C ofandida the irrigated contents sp.) infectionweekly coming with of the from thecontents cavitychlorhexidine (not coming shown) gluconate from was the 0.12% cavitydetected oral (not solution through shown). During cytological was treatment,detected examination througha candida ( cCytologicalandida and promptlysp. ) examinationinfection treated of the and with itraconazolecontents capsules coming from (1 daily the cavity for 10 (not days). shown) After was nine detected months, through a considerable cytological decrease examination of the and lesion promptly treated with× itraconazole capsules (1× daily for 10 days). After nine months, a considerable wasdecrease observedpromptly of ontreatedthe panoramic lesion with was itraconazole radiography observed capsule on (notpanoramics shown).(1× daily radiography for Excision 10 days). of (not theAfter cysticshown) nine lesion,months,. Excision as a considerable well of asthe extraction cystic of thelesiondecrease impacted, as well of the mandibular as lesionextraction was right observedof the second impacted on molar,panoramic mandibular was radiography then performed.right second(not shown) Histopathologicalmolar. E, xcisionwas then of theperformed. examination cystic confirmedHistopathologicallesion, the as well diagnosis as examination extraction of a dentigerous of confirmedthe impacted cyst the (Figure mandibular diagnosis4). One ofright a year dentigerous second later, molar a new cyst, was panoramic (Figure then performed.4). radiographyOne year Histopathological examination confirmed the diagnosis of a dentigerous cyst (Figure 4). One year waslater, carried a new out panoramic showing a radiography complete mandible was carried bone out healing showing (Figure a complete5). The mandible mandibular bone right healing third later, a new panoramic radiography was carried out showing a complete mandible bone healing molar(Figure was 5). only The removed mandibular after right its migration third molar to was the retromolaronly removed region after in its order migration to cause to the the retromolar least surgical (Figure 5). The mandibular right third molar was only removed after its migration to the retromolar region in order to cause the least surgical trauma (Figure 5). traumaregion (Figure in order5). to cause the least surgical trauma (Figure 5).

Figure 3. Sagital multi-slice computed tomography (MSCT) image: A large uniloculated lesion involving impacted mandibular right second molar. The lesion attenuation coeﬃcient varies from 3.9 to 22.9 HU.

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Figure 3. Sagital multi--sliceslice computedcomputed tomographytomography ((MSCT) imageimage:: A largelarge uniloculateduniloculated lesionlesion Dent. J. 2019involvinginvolving, 7, 76 impactedimpacted mandibular rightright second second molar. molar. T The lesionlesion attenuation coefficient 4 of 7 varies from 3.9 to 22.9 HU.

FigureFigure 4. Photomicrograph: 4. Photomicrograph A non-keratinizing:: A non--keratinizing epithelial epithelial lining withoutlining without rete pegs rete and pegs a ﬁbrous and walla withfibrousfibrous rare inﬂammatory wallwall with rarerare cells. inflammatoryinflammatory HES, 20. cellscells.. HES, ×20. ×

FigureFigure 5.5. PanoramicPanoramic radiographyradiography radiography one one year year later later::: A completecomplete mandiblemandible bone bone healing. healing.

3. Discussion3. Discussion and and Conclusions Conclusions AlthoughAlthough dentigerous dentigerous cysts cysts oftenoften appear appear as as a asimple simple radiolucentradiolucent radiolucent areaarea area surroundingsurrounding surrounding thethe crown the crown of of anan impactedimpactedimpacted tooth, tooth, largelarge cysts cysts may may show show a a multilocular multilocular featurefeature feature andand and sugsug suggestgest other other lesions lesions such such as as ameloblastomaameloblastoma and and OKC OKC [2 ,[23[2].,3,3 Therefore,].]. Therefore, a histologicala histologicalistological examination examinationexamination is isis essential essential in inin order orderorder to toto precise precise the diagnosisthethe diagnosis [1,11]. Dentigerous[1,11[1,11]].. Dentigerous cysts present cysts present a cavity a lined cavity by lined a non-keratinized by a non--keratinized stratified stratified epithelium containingepithelium between containing two between and three twotwo layers and threethree of cuboidal layerslayers ofof and cuboidal/or flattened and/orcells. flattenedflattened The cells.cells. connective The connective tissue wall is usuallytissuetissue wall fibrous isis usually and oftenfibrousfibrous devoid andand often of inflammatorydevoid of inflammatory cells. Odontogenic cells. Odontogenic keratocysts keratocyst shows show a cavity lineda cavity by a thinlinedlined and by by regulara thin and parakeratinized regular parakeratinized stratified squamousstratified squamous epithelium epithelium without with rateout pegs. rate The pegs. The basal layer cells are cuboidal or columnar and are often hyperchromatic. The lumen of these basal layer cells are cuboidal or columnar and are often hyperchromatic. The lumen of these cysts cysts contains cheese--likelike material representingrepresenting aggregated keratin scales.. Similar to thethe dentigerousdentigerous contains cheese-like material representing aggregated keratin scales. Similar to the dentigerous cyst, cyst, the interface between OKC’’s epithelium and connective tissue is flat.. Unicystic ameloblastomas the interface between OKC’s epithelium and connective tissue is flat. Unicystic ameloblastomas are a are a variant of the ameloblastoma which appear as cysts.. The epithelium lining these cysts,, however,however, variant of the ameloblastoma which appear as cysts. The epithelium lining these cysts, however, is isis composedcomposed of ameloblastic cells showing palisading and reverse nuclear polarity. The suprabasilar composedareas often of ameloblasticdisplay a loosenloosen cells stellate showing reticulum palisading appearance and reverse.. The same nuclear histological polarity. features The suprabasilarare found areasinin thethe often solidsolid display variantvariant a loosen ofof ameloblastomasameloblastomas stellate reticulum [1,2].[1,2]. appearance. The same histological features are found in the solid variant of ameloblastomas [1,2]. Unlike dentigerous cysts, ameloblastomas and odontogenic keratocysts have an aggressive behaviour and require different forms of treatment. Ameloblastomas are benign odontogenic tumours that usually require a wide surgical resection [2,12] whereas dentigerous cysts are usually treated with enucleation and curettage. Odontogenic keratocysts are benign cysts which have a relatively high rate Dent. J. 2019, 7, 76 5 of 7 of recurrence. They are preferably treated with surgical enucleation, marsupialization, decompression or marginal resection [13,14]. We presented a case of dentigerous cyst initially diagnosed as ameloblastoma having hemi-mandibulectomy as a suggested therapy. The ameloblastoma diagnosis was given by taking only the panoramic radiography and clinical aspect into account. The multilocular feature observed in the panoramic radiography is probably due to an uneven expansion of a large dentigerous cyst in areas of different bone densities [2]. It is known that panoramic radiography is a useful aid for diagnoses, but it only shows two-dimensional images of three-dimensional structures. In addition, it has a limited value to determine the lesion size and margins, tissue composition, as well as bone destruction and expansion patterns [15]. In our study, MSCT was carried out and, unlike the panoramic radiography, it showed an area of hypoattenuation without septations (Figure3). We also found a variation of the tissue attenuation coefficient from 3.9 to 22.9 HU, which suggested a lesion containing serous fluid compatible with the content of a dentigerous cyst [16]. According to the literature, ameloblastomas (solid/multicystic type) have 35.9 plus/minus 12.6 HU and the OKCs show 28.4 plus/minus 10.5 [17] or up to 40 HU [14]. Unicystic ameloblastomas present a Hounsfield Unit of 31.0 plus/minus 6.0 HU [17]. It is known that MSCT provides accurate measurement of the tissue attenuation coefficient [7–10]. In addition, the incisional biopsy done during the marsupialization confirmed the diagnosis of dentigerous cyst [1,11]. Because of the lesion extension [11], the marsupialization was the initial treatment of choice. Decompression and marsupialization are both conservative procedures aimed at reducing the cyst size without damaging anatomical structures such as alveolar nerve, or even causing mandibular fracture during the surgery. In addition, these techniques allow a gradual apposition of bone tissue before the complete cyst enucleation [18–21]. Through cytological examination, Candida sp. infection coming from the cavity was observed and promptly treated with itraconazole (1 daily for 10 days). The cytological smear is used in clinical × practice [22] and may be useful during the marsupialization treatment. After a considerable decrease nine months later, the lesion was excised, and the impacted mandibular right second molar was extracted. According to Anavi et al. [19], the average of dentigerous cyst decompression is 7.6 months in patients under 18 years of age. In children, only the marsupialization, decompression and/or enucleation of the dentigerous cyst have been suggested in an attempt to preserve the tooth and promote its eruption [23–25]. In our case, preserving the tooth was not considered possible, mainly due to the size of the cystic lesion. Histological analysis confirmed the diagnosis of a dentigerous cyst which presented a wall of uninflamed fibrous connective tissue lined by a thin non-keratinized stratified epithelium. In case of large or radiographically unusual cystic lesions, it is mandatory to perform a biopsy before planning surgical treatment. Depending on lesion size and diagnosis, an enucleation, marsupialization or decompression could be the forms of treatment chosen. In our case, the initial incisional biopsy made during the marsupialization process provided the diagnosis of a dentigerous cyst allowing a conservative approach. In addition, the tissue attenuation coefficient analysis done by MSCT was also useful in the diagnosis process to eliminate the possibility of other lesions such as a solid ameloblastoma or an odontogenic keratocyst.

Author Contributions: C.P.M.-K. and C.M.: cytological and histopathological diagnoses; T.L., C.R.M. and C.P.M.-K.: conception and drafting of the manuscript; C.R.M. and H.R.M.: patient care and interpretation of the MSCT-scan; C.M., T.L., C.P.M.-K., C.R.M. and H.R.M.: critical revision of the manuscript. Funding: Not applicable. Conflicts of Interest: The authors declare that there is no conflict of interest regarding the publication of this paper. Ethics Statement: This case report is for academic communication only. It was carried out in accordance with The Code of Ethics of the World Medical Association (Declaration of Helsinki). Written consent was obtained from a family member and every precaution was taken to protect the privacy of the patient and the confidentiality of their personal information. Dent. J. 2019, 7, 76 6 of 7

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