AbstractAn adenomatoid tumour was found in the anteriormaxillary region of a 15 year old female patient.Two impacted teeth were found in the tumour. Thelateral incisor found in the tumour was dilacerated,and the roots of the first premolar were resorbed. Areview of the English literature indicated that 294similar cases have been reported.Key words: Adenomatoid odontogenic tumour, casereport.(Received for publication March 1994. RevisedDecember 1994. Accepted January 1995.)

IntroductionThe adenomatoid odontogenic tumour is a rare

tumour that comprises only 0.1 per cent of tumoursand cysts of the jaw and 3 per cent of all odontogenictumours.1 A most comprehensive review of theodontogenic adenomatoid tumour was carried outin 1970 by Giansanti and colleagues2 who surveyedthree cases. In 1975, Courtney and Kerr3 reported20 additional cases. In 1981 Stroncek et al.4 exam-ined 37 cases reported in the English literature. Inaddition, in 1990, Toida et al.5 r e v i e wed 126Japanese cases. In all, 294 cases in the literature werereviewed. The lesion is most frequently encounteredin the second decade of life,1-20 with 19 years beingthe mean of the cases reviewed. A range of ages from36 to 82 years has been reported in the literature.

Australian Dental Journal 1997;42:(5):315-8

Adenomatoid odontogenic tumour(adenoameloblastoma). Case report and review of theliterature

Ertun Day, DDS, PhD*Gelengul Gurbuz, DDSO. Murat Bilge, DDS, PhDM. Akif iftcioglu, MD, PhD

The tumour affects females more than males inalmost a two to one ratio.2-5 The maxilla is involvednearly twice as frequently as the mandible.2-5,9,10

Unerupted permanent teeth were associated withthis lesion in one-third of the cases.2-5 In a few cases,more than one unerupted tooth was associated withthe tumour.4,5

The cuspid is the tooth most commonly associat e dwith the adenomatoid odontogenic tumour.4 , 5 , 8 , 1 1

Three-quarters of the tumours involved the anterioraspect of the jaws, particularly the incisor-canine-premolar region, of which the canine region is themost common site.3-5,8 The lesion usually appearsradiographically as a unilocular lesion but at leastfour cases of multilocular appearance have beenreported.2,4,8 Radiopacities are often seen in the peri-coronal radiolucency. This phenomenon occurred in65 per cent of the cases Giansanti et al.2 reviewed inwhich radiographs were available, or where mentionof the radiographic appearance was made. In onecase, irregular root resorption,11 and in two casesdilaceration, were reported.10,14 The size of the lesionusually varied from 15 to 30 mm in diameter.Several larger tumours have been noted, the largestwas more than 120 mm.10 Radiographically, thelesion frequently looked like a dentigerous cyst orfollicular cyst.1-20 The radiolucency associated withthe odontogenic adenomatoid tumour may extendmore apically than the dentigerous cyst.3 An intra-oral or extraoral swelling was the main symptom,and the swelling was usually painless and slowgrowing.5,7,13

Case reportA 15 year old girl presented with a swelling in the

lateral-canine area of upper right jaw. She had visited

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*Assistant Professor, Department of Oral and Maxillofacial Surgery,Faculty of Dentistry, Ataturk Unive rs i t y, Tu r k e y.Researcher, Department of Oral and Maxillofacial Surgery, Fa c u l t yof Dentistry, Ataturk Unive rs i t y, Tu r k e y. A s s o c i ate Professor and Chairman, Department of Oral Diagnosisand Radiology, Faculty of Dentistry, Ataturk Unive rs i t y, Tu r k e y.Assistant Professor, Department of Pat h o l o g y, School of Medicine,A t aturk Unive rs i t y, Tu r k e y.

In the root of the lateral incisor in the lesion,dilaceration was seen (Fig. 2). There was irregularroot resorption in the right maxillary first premolar.The lesion was directly associated with both the noseand sinus. Aspiration was attempted and yielded 5mL of a turbid grey-tan fluid. Under localanaesthesia the lesion and impacted lateral incisorand canine were extracted.

After the operation the specimen was fixed in 10per cent formal saline and prepared for histologicale x a m i n ation. Some sections were stained withhaematoxylin-eosin, while others were stained withCongo red and crystal violet.

Microscopically a poor connective tissue stromawas seen. Spindle or polyhedral epithelial cells inthis stroma displayed duct-like structures. In someareas amorphous eosinophilic material was seenamong the tumour cells in the form of solid nests.This material showed a positive reaction with theCongo red dye, but with the crystal violet the reactionwas negative. Microcalcifications were seen in allregions of the tumour (Figs. 3-6).

DiscussionIt was Stafne who identified the adenomatoid

odontogenic tumour for the first time in 1948.2,4,9,10

S u b s e q u e n t l y, in cases reported by va rious authors4 - 6

these tumours are described as intraoral-extraoralswellings in the maxilla2,5,9,10 which generally aremore frequently seen in females2-5 and which mostlyoccur in the second decade of life.1-20 Apart from afew exceptional cases4,5 the tumour is associated withunerupted teeth. The unerupted teeth are usuallycanine or lateral incisors.2-5,8 Irregular root resorptionand dilaceration within the lesion are only infrequentlyreported in the literature.10,14 Clinical, radiographicand macroscopic findings in the present case areconsistent with descriptions of the lesion in thedental literature. It was also observed that the

316 Australian Dental Journal 1997;42:5.

a dentist a year ago. The dentist without having aradiograph had started the treatment by extractingher upper deciduous lateral incisor and cuspid teethand by applying antibiotic medication. On realizingthat the swelling was getting worse, the patientpresented at the Oral Diagnosis and RadiologyClinic of the Dentistry Faculty, Ataturk University inApril 1993.

Extraoral examination disclosed a swelling in theanterior maxilla with no pain. When examined intra-orally it was seen that the right maxillary lateralincisor and canine teeth were missing and there wasa hyperaemic swelling in the vestibule.

On radiological examination (intraoral panoramicradiography, Waters sinus occlusal periapical views)a radiolucent lesion with a regular border was seenin the area ranging between the right maxillarycentral and right first molar. The lesion wa s45 40 mm in size and was unilocular and showedsmall radiopaque calcification points. In the lesion,the maxillary canine was impacted in the apex offirst molar tooth and lateral incisor (Fig. 1).

Fig. 1.Intraoral radiograph showing irregular root resorption in borders of the lesion and in first premolar.Small radiopaque calcifications can be seen in the lesion.

Fig. 2.Dilaceration in root of lateral incisor in tumour, and caninedisplaced in tumour. Bar=5 mm.

5 mm

present tumour was associated with two uneruptedteeth and that there was resorption in the firstpremolar and dilaceration of the lateral incisor.According to Giansanti et al.2 after local curettage ofthe tumour a number of cases were followed up forperiods ranging from one to ten years with noreported cases of recurrence. Indeed, Giansanti etal.2 reported that the adenomatoid odontogenictumour was a completely benign tumour whichn e ver recurred once removed. In the one ye a rfollow-up in the present case no recurrence wasreported.

References01. Khan MY, Kwee H, Schneider LC, Saber I. Adenomat o i d

odontogenic tumor resembling a globu l o m a x i l l a ry cyst: light andelectron microscopic studies. J Oral Surg 1977;35:739-42.

02. Giansanti JS, Someren A, Waldron CA. Odontogenic adeno-m atoid tumors (adenoameloblastoma). Survey of 3 cases. OralSurg Oral Med Oral Pathol 1970;30:69-88.

03. Courtney RM, Kerr DA. The odontogenic adenomatoid tumor.A comprehensive study of twenty new cases. Oral Surg Oral MedOral Pathol 1975;39:424-35.

04. Stroncek GG, Acevedo A, Higa LH. An atypical odontogenicadenomatoid tumor and review of the literature. J Oral Med1981;36:102-6.

05. Toida M, Hytodo I, Okuda T, et al. Adenomatoid odontogenictumor: report of two cases and survey of 126 cases in Japan. JOral Maxillofac Surg 1990;48:404-8.

06. Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors:analysis of 706 cases. J Oral Surg 1978;36:771-8.

07. Hacihanefioglu U. The adenomatoid odontogenic tumor. OralSurg Oral Med Oral Pathol 1974;32:65-73.

08. M e yer I, Giunta JL. Adenomatoid odontogenic tumor(adenoameloblastoma): report of case. J Oral Surg 1974;32:448-51.

09. Seymour RL, Funke FW, Irby WB. Adenoameloblastoma.Report of a case and review of the literature. Oral Surg Oral MedOral Pathol 1974;38:860-5.

10. Tsaknis PJ, Carpenter WM, Shade NL. Odontogenic adeno-matoid tumor: report of case and review of the literature. J OralSurg 1977;35:146-9.

11. Nomura M, Tanimoto K, Takata T, et al. Mandibular adeno-m atoid odontogenic tumor with unusual clinicopat h o l o gi cfeatures. J Oral Maxillofac Surg 1992;50:282-5.

12. Tajima Y, Sakamoto E, Yamamoto Y. Odontogenic cyst givingrise to an adenomatoid odontogenic tumor: report of a case withpeculiar features. J Oral Maxillofac Surg 1992;50:1990-3.

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Fig. 3.Calcification and duct-like structures. H&E. 40.Fig. 4.Duct-like and adenoid structures. H&E. 100.

Fig. 5.Tumour stroma and duct-like structures. H&E. 100.Fig. 6.Amyloid-like mid-substance. H&E. 100.

3 4

5 6

13. Milobsky L, Milobsky SA, Miller GM. Adenomatoid odonto-genic tumor (adenoameloblastoma). Report of a case. Oral SurgOral Med Oral Pathol 1975-40:681-5.

14. Ebling H, Barbachan JJD. Adenoameloblastoma. Report of acase. Oral Surg Oral Med Oral Pathol 1968;26:674-8.

15. Bhaskar SN. Oral pat h o l o g y. 3rd edn. St Louis: M o s b y,1969:231-5.

16. Zegarelli EV, Kutscher AH, Hyman GA. Diagnosis of diseases ofthe mouth and jaws. 2nd edn. Philadelphia: Lea & Febiger,1978:278.

17. Stafne EC. Oral roentgenographic diagnosis; including anappendix on roentgenographic technique. 3rd edn. Philadelphia:Saunders, 1969:172-3.

18. Shafer WG, Hine BM, Levy BM. A textbook of oral pathology.4th edn. Philadelphia: Saunders, 1983:289-91.

19. Goaz PW, White LS. Oral radiology. 2nd edn. St Louis: Mosby,1987:525-33.

20. Harorl A. Dis Hekimligi Radyolojisi. Erzurum: Atatu r kUniversity, 1992. Y. No. 742:209-11.

Address for correspondence/reprints:Dr O. M. Bilge,

Department of Oral Diagnosis and Radiology,Faculty of Dentistry,

Ataturk University,Erzurum,

Turkey.

318 ustralian Dental Journal 1997;42:5.