Swelling of the upper lip can result from various diseases such as salivary tumors, infectious and inflammatory diseases and cysts. Among the latter, dentigerous cysts, typically involving unerupted teeth, are sometimes associated with supernumerary teeth in the maxillary anterior incisors region called the mesiodens. We report an unusual case of a large dentigerous cyst associated with an impacted mesiodens in a 42-year-old male who presented with a slow-growing swelling in the upper li

Lombardi, Tommaso

Richter, Michel

Scolozzi, Paolo

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MISCELLANEOUSPaolo Scolozzi Æ Tommaso Lombardi Æ Michel RichterUpper lip swelling caused by a large dentigerous cystReceived: 22 December 2003 / Accepted: 9 February 2004 / Published online: 5 May 2004 Springer-Verlag 2004Abstract Swelling of the upper lip can result from variousdiseases such as salivary tumors, infectious and inﬂam-matory diseases and cysts. Among the latter, dentigerouscysts, typically involving unerupted teeth, are sometimesassociated with supernumerary teeth in the maxillaryanterior incisors region called the mesiodens. We reportan unusual case of a large dentigerous cyst associatedwith an impacted mesiodens in a 42-year-old male whopresented with a slow-growing swelling in the upper lip.Keywords Upper lip Æ Dentigerous cyst Æ MesiodensIntroductionDentigerous cysts, also known as follicular cysts, aredevelopmental cysts formed by ﬂuid accumulation be-tween the reduced enamel epithelium and the enamelsurface. They surround the crown of an unerupted tooth,most often the mandibular third molars and maxillarycanines. They are occasionally associated with supernu-merary teeth. First named by Bolk in 1917, mesiodentesare the most common supernumerary teeth [4]. They aretypically found in the midline of the anterior maxilla, inthe palate area between the apical and cervical regions ofthe permanent incisors, and most of them remain im-pacted. The incidence of mesiodentes as reported in theliterature ranges from 0.15 to 4 percent [16]. Althoughthe permanent dentition is most often aﬀected, deciduoussupernumerary teeth are occasionally reported.We report a case of a relatively large dentigerous cystassociated with an unerupted mesiodens in a 42-year-oldmale who presented with a slow-growing mass in theupper lip. The cyst was treated by enucleation andimmediate autogenous cancellous bone graft.Case reportIn October 2001, a 42-year-old Ethiopian male was re-ferred to our Maxillofacial Unit by the ENT Depart-ment for investigation of a slow-growing mass on theupper lip. The mass had been present for several yearsand resulted in malpositioning of the central maxillaryincisors. Intraoral examination revealed a soft, labialcyst-like swelling measuring 3.5·2.0 cm (Fig. 1). Max-illary anterior teeth were vital and not sensitive to per-cussion, except for the right central incisor. A panoramicradiograph revealed a relatively large and well-deﬁnedradiolucency extending from the right canine to the leftlateral maxillary incisor, enveloping an unerupted me-siodens. Computerized tomography showed a cystic le-sion measuring 3.4 cm horizontally, 2.5 cm verticallyand 2.3 cm sagittally, with the labial and palatal corticalbone expanded and eroded (Fig. 2). The right maxillarycanine, the right lateral and central incisors and the leftcentral and lateral incisors were treated endodonticallythe day before the operation because of their intimaterelationship with the lesion. Under general anesthesia,the cyst was wholly enucleated together with the une-rupted mesiodens (Fig. 3). The bony defect was ﬁlledwith autogenous cancellous iliac bone graft. The post-operative course was uneventful.The gross specimen consisted of a brown cyst mea-suring 2.7·0.4·2.0 cm with a small monoradicularP. Scolozzi (&)Department of Otolaryngology and Head and Neck Surgery,Division of Oral and Maxillofacial Surgery,CHUV (Centre Hospitalier Universitaire Vaudois),1011 Lausanne, SwitzerlandE-mail: scolozzi@hotmail.comTel.: +41-213142731Fax: +41-213141347T. LombardiLaboratory of Oral Histopathology, Faculty of Medicine,19 Rue Barthe´lemy-Menn, 1205 Geneva, SwitzerlandM. RichterDivision of Reconstructive Surgery Oral and Maxillofacial SurgeryUnit, University of Geneva Hospital,24 Micheli-du-Crest, Geneva, SwitzerlandEur Arch Otorhinolaryngol (2005) 262: 246–249DOI 10.1007/s00405-004-0782-4malformed and dwarfed supernumerary tooth. The cystwas inserted on the cervical zone of the tooth. Micro-scopic examination showed a cyst lined by an epitheliumwith cuboidal and columnar cells resting on a ﬂat basallamina. In focal areas, non-keratinizing stratiﬁed squa-mous and transitional epithelium was also observed(Fig. 4). The connective tissue wall consisted of ﬁbroustissue without inﬂammatory inﬁltrate. The ﬁnal diag-nosis was that of a dentigerous cyst associated withmesiodens.DiscussionSwelling of the upper lip can result from diﬀerent dis-eases ranging from infectious diseases (abscesses orcellulites), allergic diseases (angioedema), neoplasms,especially of salivary origin (canalicular adenoma,pleomorphic adenoma), granulomatous diseases (iso-lated Miescher’s granulomatous cheilitis or as part ofMelkersson-Rosenthal syndrome, Crohn’s disease andsarcoidosis), inﬂammatory diseases (cheilitis glandularis)and cysts (epidermoid or epidermal inclusion cyst,nasolabial cyst, agressive odontogenic cyst such as ker-atocysts extending through the bone cortex and denti-gerous cysts).Dentigerous cysts—typically associated with im-pacted teeth—are sometimes associated with supernu-merary teeth in the maxillary anterior incisors regioncalled the mesiodens. The most common pathologicalﬁndings reported to be associated with mesiodens areretention of adjacent incisors, malposition and diastemaformation [16, 2]. Contrarily, development of a denti-gerous cyst on an impacted mesiodens is relatively rareFig. 2 Sagittal CT image showing the mesiodens impacted withinthe palatal bone. The labial cortical bone is completely eroded bythe cystFig. 3 Intraoperative view showing the cyst bulging from theanterior maxillaFig. 4 Microscopic examination showing a portion of the cyst linedby an epithelium with cuboidal and columnar cells resting on a ﬂatbasal lamina. Inset shows an area of the cyst lined by nonkerati-nizing stratiﬁed squamous epithelium. (hematoxylin-eosin, originalmagniﬁcation ·20)Fig. 1 Intraoral view showing a labial cyst-like swelling247[16]. Stafne [15] ﬁrst described the association of denti-gerous cyst with the supernumerary maxillary teeth andfound an incidence of 5.5% among 180 patients with 200supernumerary teeth occurring in the region of the uppercentral incisors. Bodin reviewed 350 supernumeraryteeth in 290 patients and found a widened pericoronalspace in 12% of cases, a rather high frequency thatprobably indicates that the risk of cyst formation withsupernumerary teeth should not be disregarded [2].Primosch reported an enlarged dental sac in 30% ofcases, but a histological diagnosis of dentigerous cyst inonly 4 to 9% of cases [13]. Recently, von Arx clinicallyand radiologically reviewed 113 mesiodentes in 90 pa-tients and found a follicular cyst (pericoronal space>5 mm on the conventional radiograph) in only threepatients [16]. Lustmann reviewed the literature from1900–1988 and found 42 cases of dentigerous cystassociated with supernumerary teeth [9].In our review of the literature, we have found onlyeight cases of large dentigerous cysts (>3 cm) associatedwith a mesiodens (Table 1), and none was treated bybone graft. While some authors propose enucleationwithout using bone grafting for treating large mandib-ular cysts [5], enucleation followed by immediate bonegrafting procedure is the recommended treatment forlarge jaw cysts with supernumerary or wisdom teeth [3,14]. Enucleation guarantees the complete removal ofthe lesion and provides the entire cyst for the patho-logical examination. Diﬀerent grafting materials havebeen used to ﬁll the residual cavity, including allo-plastic bone, allogenic bone or xenogenic bone or acombination of these materials, but autogenous can-cellous grafts produce the most favorable and predict-able results, both experimentally and clinically [3, 14].They also possess excellent space maintaining proper-ties, a high concentration of osteoinductive cells andact as an osteoconductive scaﬀold during new boneformation. The iliac crest provides large amounts ofcancellous bone and therefore is the site of choice toharvest graft, even though this has the disadvantageof involving a second surgical site procedure withincreased morbidity [3, 14].Table 1 Large dentigerous cysts (>3 cm) associated with impacted mesiodensAuthors Gender Age(years)Localization Duration Symptoms Size (cm) TherapyStafne(1931) [15]– – Palate – – Entire palate –Haag, Frankand Nicolas(1966) [7]F 13 Between the rightcentral incisor andthe left canine15 days Left hemifacial painand swelling of theleft paranasal region3·3.5 cm EnucleationCiola andCatena(1972) [6]M 46 Apical to themaxillary anteriorteeth betweenthe two canines– ‘‘Front teeth sensitiveto cold air.’’Large EnucleationArcher(1975) [1]1) F 12 Between from leftcentral incisor andthe ﬁrst right molar.– – Large Marsupialization2) – – Between the twocanines– – Large MarsupializationPapadopoulos(1981) [12]F 40 From the upperright central incisorto the upper leftsecond premolar10 years Repeated swellingover the upper leftcentral and lateralincisor region(buccal and palatal)3·2x2 cm EnucleationMost and Roy(1982) [10]M 30 Entire right antrumand one half ofthe left antrum– Pain and swellingof the right maxillarymucogingival fold area9·1.5·0.8 cm EnucleationLustmannand Bodner(1988) [9]1) M 37 Premaxilla 6 days Acute painful swellingof the premaxillaryregion. AsymptomaticEntirepremaxillaEnucleation2) F 71 Premaxilla – Large EnucleationNoor Awangand Huat Siar(1989) [11]1) M 34 From the maxillarymidline to the upperleft ﬁrst premolar1 month Swelling in the leftpremaxilla.3·2 cm labially Enucleation2·2 cm (palate)2) F 24 From upper rightcanine to the upperleft central incisor– Swelling obliteratingthe upper labial sulcus3·3cm EnucleationPresent case M 42 Premaxilla SeveralyearsMalposition of thecentral incisors andswelling of theupper lip3.4·2.5·2.3 cm Enucleationand autogenouscancellous bonegraft from iliaccrest248In our opinion, marsupialization should be limited tothose particular cases in which teeth have to be con-served and brought into the dental arch or in an un-healthy or debilitated patient.In conclusion, large dentigerous cysts arising fromunerupted mesiodens should be kept in mind in thediﬀerential diagnosis of upper lip swelling, particularly ifassociated with dental anomalies of the maxillary inci-sors such as malposition and diastema. To prevent thedevelopment of a dentigerous cyst and to avoid un-wanted eﬀects on neighboring teeth, supernumeraryanterior teeth should be removed surgically or observedwith regular radiographic controls [8].References1. Archer WH (1975) Oral and maxillofacial surgery, 5th edition.WB Saunders, Philadelphia, pp 590–5962. Bodin I, Julin P, Thomsson M (1981) Hyperdontia III.Supernumerary anterior teeth. Dentomaxillofac Radiol10:35–413. Bodner L (1996) Eﬀect of decalciﬁed freeze-dried bone allografton the healing of jaw defects after cyst enucleation. J OralMaxillofac Surg 54:1282–12864. Bolk L (1917) Die u¨berza¨hligen oberen Incisivi des Menschen.Dtsch Mschr Zahnheilk 35:1855. Chiapasco M, Rossi A, Motta JJ, Crescentini M (2000)Spontaneuous bone regeneration after enucleation of largemandibular cysts. A radiographic computed analysis of 27consecutive cases. J Oral Maxillofac Surg 58:942–9486. Ciola B, Lorne Catena D (1972) Midline maxillary cyst com-plicated by unerupted mesiodens. Oral Surg 34:978–9837. Haag R, Frank RM, Nicolas P (1966) Kyste corono-dentairesur me´siodens inversle´e. Rev Stomatol 67:217–2228. Koch H, Schwartz O, Klausen B (1986) Indications for surgicalremoval of supernumerary teeth in the premaxilla. Int J OralMaxillofac Surg 15:273–2819. Lustmann J, Bodner L (1988) Dentigerous cysts associated withsupernumerary teeth. Int J Oral Maxillofac Surg 17:100–10210. Most DS, Roy EP (1982) A large dentigerous cyst associatedwith a supernumerary tooth. J Oral Maxillofac Surg 40:119–12011. Noor Awang M, Chong Huat S (1989) Dentigerous cyst due tomesiodens: report of two cases. J Ir Dent Assoc 35:117–11812. Papadopoulos MS (1981) An unusual case of dentigerous cystdue to a mesiodens. Br J Oral Surg 19:307–30813. Primosch RE (1981) Anterior supernumerary teeth-assessmentand surgical intervention in children. Pediatr Dent 3:204–21514. Richter M, Laurent F, Chausse JM (1986) Homologouscancellous bone graft for large jaw defect caused by bone cysts.J Oral Maxillofac Surg 44:447–45315. Stafne EC (1931) Supernumerary upper central incisor. DentCosmos 73:976–98016. von Arx T (1992) Anterior maxillary supernumerary teeth: aclinical and radiographic study. Aust Dent J 3:189–195249

Upper lip swelling caused by a large dentigerous cyst

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Upper lip swelling caused by a large dentigerous cyst

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