Adenomatoid odontogenic tumour adenoameloblastoma Case report

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

Adenomatoid odontogenic tumour
(adenoameloblastoma). Case report and review of the
Ertunç Dayı, DDS, PhD*
      u u u
Geleng¨ l G¨ rb¨ z, DDS†
O. Murat Bilge, DDS, PhD‡
M. Akif Çiftcioˇ lu, MD, PhD§

   Abstract                                                              The tumour affects females more than males in
   An adenomatoid tumour was found in the anterior                    almost a two to one ratio.2-5 The maxilla is involved
   maxillary region of a 15 year old female patient.                  nearly twice as frequently as the mandible.2-5,9,10
   Two impacted teeth were found in the tumour. The                   Unerupted permanent teeth were associated with
   lateral incisor found in the tumour was dilacerated,               this lesion in one-third of the cases.2-5 In a few cases,
   and the roots of the first premolar were resorbed. A               more than one unerupted tooth was associated with
   review of the English literature indicated that 294
   similar cases have been reported.                                  the tumour.4,5
   Key words: Adenomatoid odontogenic tumour, case
                                                                         The cuspid is the tooth most commonly associated
   report.                                                            with the adenomatoid odontogenic tumour.4,5,8,11
   (Received for publication March 1994.              Revised
                                                                      Three-quarters of the tumours involved the anterior
   December 1994. Accepted January 1995.)                             aspect of the jaws, particularly the incisor-canine-
                                                                      premolar region, of which the canine region is the
                                                                      most common site.3-5,8 The lesion usually appears
Introduction                                                          radiographically as a unilocular lesion but at least
  ‘The adenomatoid odontogenic tumour is a rare                       four cases of multilocular appearance have been
tumour that comprises only 0.1 per cent of tumours                    reported. 2,4,8 Radiopacities are often seen in the peri-
and cysts of the jaw and 3 per cent of all odontogenic                coronal radiolucency. This phenomenon occurred in
tumours’.1 A most comprehensive review of the                         65 per cent of the cases Giansanti et al. 2 reviewed in
odontogenic adenomatoid tumour was carried out                        which radiographs were available, or where mention
in 1970 by Giansanti and colleagues2 who sur veyed                    of the radiographic appearance was made. In one
three cases. In 1975, Courtney and Kerr3 reported                     case, irregular root resorption,11 and in two cases
20 additional cases. In 1981 Stroncek et al.4 exam-                   dilaceration, were reported.10,14 The size of the lesion
ined 37 cases reported in the English literature. In                  usually varied from 15 to 30 mm in diameter.
addition, in 1990, Toida et al.5 reviewed 126                         Several larger tumours have been noted, the largest
Japanese cases. In all, 294 cases in the literature were              was more than 120 mm.10 Radiographically, the
reviewed. The lesion is most frequently encountered                   lesion frequently looked like a dentigerous cyst or
in the second decade of life,1-20 with 19 years being                 follicular cyst.1-20 The radiolucency associated with
the mean of the cases reviewed. A range of ages from                  the odontogenic adenomatoid tumour may extend
36 to 82 years has been reported in the literature.                   more apically than the dentigerous cyst.3 An intra-
                                                                      oral or extraoral swelling was the main symptom,
*Assistant Professor, Department of Oral and Maxillofacial Surgery,
                                                                      and the swelling was usually painless and slow
Faculty of Dentistry, Atat¨ rk University, Turkey.
                          u                                           growing.5,7,13
†Researcher, Department of Oral and Maxillofacial Surgery, Faculty
of Dentistry, Atat¨ rk University, Turkey.
‡Associate Professor and Chairman, Department of Oral Diagnosis       Case report
and Radiology, Faculty of Dentistry, Atat¨ rk University, Turkey.
§Assistant Professor, Department of Pathology, School of Medicine,
                                                                         A 15 year old girl presented with a swelling in the
Atat¨ rk University, Turkey.
    u                                                                 lateral-canine area of upper right jaw. She had visited
Australian Dental Journal 1997;42:5.                                                                                                 315
                  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.

a dentist a year ago. The dentist without having a                             In the root of the lateral incisor in the lesion,
radiograph had started the treatment by extracting                          dilaceration was seen (Fig. 2). There was irregular
her upper deciduous lateral incisor and cuspid teeth                        root resorption in the right maxillary first premolar.
and by applying antibiotic medication. On realizing                         The lesion was directly associated with both the nose
that the swelling was getting worse, the patient                            and sinus. Aspiration was attempted and yielded 5
presented at the Oral Diagnosis and Radiology                               mL of a turbid grey-tan fluid. Under local
Clinic of the Dentistry Faculty, Atat¨ rk University in                     anaesthesia the lesion and impacted lateral incisor
April 1993.                                                                 and canine were extracted.
   Extraoral examination disclosed a swelling in the                           After the operation the specimen was fixed in 10
anterior maxilla with no pain. When examined intra-                         per cent formal saline and prepared for histological
orally it was seen that the right maxillary lateral                         examination. Some sections were stained with
incisor and canine teeth were missing and there was                         haematoxylin-eosin, while others were stained with
a hyperaemic swelling in the vestibule.                                     Congo red and crystal violet.
   On radiological examination (intraoral panoramic                            Microscopically a poor connective tissue stroma
radiography, Waters sinus occlusal periapical views)                        was seen. Spindle or polyhedral epithelial cells in
a radiolucent lesion with a regular border was seen                         this stroma displayed duct-like structures. In some
in the area ranging between the right maxillary                             areas amorphous eosinophilic material was seen
central and right first molar. The lesion was                               among the tumour cells in the form of solid nests.
45 40 mm in size and was unilocular and showed                              This material showed a positive reaction with the
small radiopaque calcification points. In the lesion,                       Congo red dye, but with the crystal violet the reaction
the maxillary canine was impacted in the apex of                            was negative. Microcalcifications were seen in all
first molar tooth and lateral incisor (Fig. 1).                             regions of the tumour (Figs. 3-6).

                                                                              It was Stafne who identified the adenomatoid
                                                                            odontogenic tumour for the first time in 1948.2,4,9,10
                                                                            Subsequently, in cases reported by various authors4-6
                                                                            these tumours are described as intraoral-extraoral
                                                                            swellings in the maxilla2,5,9,10 which generally are
                                                                            more frequently seen in females2-5 and which mostly
                                                                            occur in the second decade of life.1-20 Apart from a
                                                                            few exceptional cases4,5 the tumour is associated with
                                                                            unerupted teeth. The unerupted teeth are usually
                                                                            canine or lateral incisors.2-5,8 Irregular root resorption
                                                                            and dilaceration within the lesion are only infrequently
                                                                            reported in the literature.10,14 Clinical, radiographic
      5 mm                                                                  and macroscopic findings in the present case are
Fig. 2.–Dilaceration in root of lateral incisor in tumour, and canine
                                                                            consistent with descriptions of the lesion in the
                  displaced in tumour. Bar=5 mm.                            dental literature. It was also observed that the
316                                                                                                             Australian Dental Journal 1997;42:5.
    3                                                                   4

    5                                                                   6

                                          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.

present tumour was associated with two unerupted                        04. Stroncek GG, Acevedo A, Higa LH. An atypical odontogenic
                                                                            adenomatoid tumor and review of the literature. J Oral Med
teeth and that there was resorption in the first                            1981;36:102-6.
premolar and dilaceration of the lateral incisor.
                                                                        05. Toida M, Hytodo I, Okuda T, et al. Adenomatoid odontogenic
According to Giansanti et al.2 after local curettage of                     tumor: report of two cases and survey of 126 cases in Japan. J
the tumour a number of cases were followed up for                           Oral Maxillofac Surg 1990;48:404-8.
periods ranging from one to ten years with no                           06. Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors:
reported cases of recurrence. Indeed, Giansanti et                          analysis of 706 cases. J Oral Surg 1978;36:771-8.
al.2 reported that the adenomatoid odontogenic                                         g
                                                                        07. Hacihanefioˇ lu U. The adenomatoid odontogenic tumor. Oral
tumour was a completely benign tumour which                                 Surg Oral Med Oral P athol 1974;32:65-73.

never recurred once removed. In the one year                            08. Meyer I, Giunta JL. Adenomatoid odontogenic tumor
                                                                            (adenoameloblastoma): report of case. J Oral Surg 1974;32:448-
follow-up in the present case no recurrence was                             51.
reported.                                                               09. Seymour RL, Funke FW, Irby WB. Adenoameloblastoma.
                                                                            Report of a case and review of the literature. Oral Surg Oral Med
                                                                            Oral Pathol 1974;38:860-5.
01. Khan MY, Kwee H, Schneider LC, Saber I. Adenomatoid                 10. Tsaknis PJ, Carpenter WM, Shade NL. Odontogenic adeno-
    odontogenic tumor resembling a globulomaxillary cyst: light and         matoid tumor: report of case and review of the literature. J Oral
    electron microscopic studies. J Oral Surg 1977;35:739-42.               Surg 1977;35:146-9.
02. Giansanti JS, Someren A, Waldron CA. Odontogenic adeno-             11. Nomura M, Tanimoto K, Takata T, et al. Mandibular adeno-
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03. Courtney RM, Kerr DA. The odontogenic adenomatoid tumor.            12. Tajima Y, Sakamoto E, Yamamoto Y. Odontogenic cyst giving
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13. Milobsky L, Milobsky SA, Miller GM. Adenomatoid odonto-          19. Goaz PW, White LS. Oral radiology. 2nd edn. St Louis: Mosby,
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318                                                                                                    ustralian Dental Journal 1997;42:5.

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