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Korean Journal of Oral and Maxillofacial Radiology 2006; 36 : 177-82



Diagnostic ability of differential diagnosis in ameloblastoma and
odontogenic keratocyst by imaging modalities and observers

Tae-In Gang, Kyung-Hoe Huh, Won-Jin Yi**, Min-Suk Heo**, Sam-Sun Lee*,
Jeong-Hwa Kim, Je-Woon Moon, Soon-Chul Choi*
Department of Oral and Maxillofacial Radiology
*Department of Oral and Maxillofacial Radiology, Dental Research Institute
**Department of Oral and Maxillofacial Radiology, Dental Research Institute, and BK21
School of Dentistry, Seoul National University

ABSTRACT

      Purpose : To evaluate the diagnostic ability in differentiating between ameloblastoma and odontogenic keratocyst
      according to the imaging modalities and observers.
      Materials and Methods : We evaluated thirty-six cases of ameloblastomas and forty-seven cases of odontogenic
      keratocysts all histologically confirmed. Six oral and maxillofacial radiologists diagnosed the lesions by 3 methods:
      using panoramic radiograph, using computed tomograph (CT), and using panoramic radiograph and CT. The
      observers were classified by 3 groups: group 1 had experienced over 10 years in oral and maxillofacial radiologic
      field, group 2 had experienced for 3-4 years, and group 3 was in the process of residentship. After over 2 weeks, the
      observers diagnosed them by the same methods.
      Results : The ROC curve areas except for group 3 were the highest with interpretation using panoramic radiograph
      and CT, followed by interpretation using CT only, and the lowest with interpretation using panoramic radiograph
      only. The overall difference was not found in diagnostic ability among groups in using panoramic radiograph only,
      but there was difference in diagnostic ability of group 1 and 2 vs 3 in using CT only, and combination panoramic
      radiograph and CT.
      Conclusions : To differentiate between ameloblastoma and odontogenic keratocyst more accurately, the
      experienced oral and maxillofacial radiologist should diagnose with combination of panoramic radiograph and CT.
      (Korean J Oral Maxillofac Radiol 2006; 36 : 177-82)

      KEY WORDS : Diagnostic Ability; Ameloblastoma; Odontogenic Keratocyst




                                                                              lingual cortical expansion is frequently present. Root resorp-
                          Introduction
                                                                              tion of adjacent teeth to the tumor is common.1
  Ameloblastomas are tumors of odontogenic epithelial                            Odontogenic keratocysts arise from cell rests of the dental
origin. Theoretically, they may arise from cell rests of the                  lamina and make up 10 to 12 percent of all developmental
enamel organ, from a developing enamel organ, from the                        odontogenic cysts.1 Histopathologically, they typically show a
epithelial lining of an odontogenic cyst, or from the basal cells             thin, friable wall, which is often difficult to enucleate from the
of the oral mucosa.1 They make up 1% of all tumors of the                     bone in one piece, and have small satellite cysts within fibrous
jaws,2 and 11% of odontogenic tumors.3 Although ameloblas-                    wall. Therefore odontogenic keratocysts often tend to recur
tomas are histopathologically benign, they have been charac-                  after treatment.7 Radiographically odontogenic keratocysts
terized as a tumor with marked tendency to recur.2,4 Radio-                   demonstrate a well-defined unilocular or multilocular radiolu-
graphically ameloblastomas show an expansile unilocular or                    cency with smooth and often corticated margins. 1 Odonto-
multilocular lesion with a “honeycomb or soap bubble”                         genic keratocysts tend to grow in and anteroposterior direction
appearance and a sharp scalloping border. 5,6 Buccal and                      within the medullary cavity of the bone without causing obvi-
                                                                              ous bone expansion.1 Displacement of teeth adjacent to the
Received October 4, 2006; accepted November 6, 2006
Correspondence to : Prof. Soon-Chul Choi                                      cyst occurs more frequently than resorption.6
Department of Oral and Maxillofacial Radiology School of Dentistry, Seoul       The surgical treatment of ameloblastomas is marginal or
National University 28, Yeongeon-dong, Jongno-gu, Seoul, 110-749, Korea
Tel) 82-2-2072-2622, Fax) 82-2-744-3919, E-mail) raychoi@snu.ac.kr            block resection, but most odontogenic keratocysts are treated

                                                                      ─ 177 ─
Diagnostic ability of differential diagnosis in ameloblastoma and odontogenic keratocyst by imaging modalities and observers


by enucleation and curretage.8,9 The preoperative differential          keratocyst. The purpose of the present study is to assess the
diagnosis between ameloblastoma and odontogenic keratocyst              diagnostic ability, intra- and interobserver agreement on
is important to establish appropriate treatment planning, but           radiologic differential diagnosis in ameloblastoma and
ameloblastoma and odontogenic keratocyst are not much                   odontogenic keratocyst by imaging modalities and observers.
different in development age or site, and sometimes they are
radiologically similiar, which makes it not easy to differenti-
                                                                                         Materials and Methods
ate between them.
   The studies of the preoperative differential diagnosis                 1. Materials
between ameloblastoma and odontogenic keratocyst have
                                                                          We examined histopathologically confirmed cases, taken
been reported over years. Tanimoto et al.10 reported that buc-
                                                                        preoperative panoramic radiograph and CT (IQ scanner,
colingual expansion of the ramus caused by odontogenic
                                                                        Picker International, USA), from January 1997 to July 2003 in
keratocysts was smaller than that caused by ameloblastomas.
                                                                        Seoul National University Dental Hospital. Thirty-six cases of
Choi11 reported that lesional outline, the root resorption of
                                                                        ameloblastomas and forty-seven cases of odontogenic kerato-
adjacent teeth, tooth displacement, and the loss of lamina dura
                                                                        cysts were selected. The cases showing the typical multilo-
could be the parameters to differentiate odontogenic kerato-
                                                                        cular characteristics of the ameloblastoma and recurrent cases
cyst and unilocular ameloblastoma. Minami et al.12 reviewed
                                                                        were not included.
the magnetic resonance findings in patients with odontogenic
keratocysts or ameloblastomas, and proposed that from the
                                                                          2. Methods
magnetic resonance (MR) findings of the walls, solid com-
ponents, and the fluid contents, odontogenic keratocysts could              1) Observers
be differentiated from ameloblastomas. Jeong13 et al. describ-            The six observers who participated in the study were
ed that maxillary lesions were more common in odontogenic               divided three groups. Group 1 was composed of the two
keratocyst, multilocular lesions were seen more frequently in           observers (observer A, B), having experience over 10 years in
ameloblastoma, and the external root resorption of adjacent             oral and maxillofacial radiologic field. Group 2 was com-
teeth showed more frequently in ameloblastoma. Tozaki et                posed of the two observers (observer C, D), having experience
al.14 reported that a rapidly enhancing area was detected in            4-5 years in oral and maxillofacial radiologic field. Group 3
cases of ameloblastoma, while no apparent rapid enhancement             was composed of the two observers (observer E, F), in the
was seen in the other cystic lesions, including odontogenic             process of residentship.
keratocyst on dynamic multislice helical computed tomograph
                                                                            2) Diagnostic method
(CT). Soh et al.15 described that the root resorption of the
                                                                          Each one of the six observers diagnosed the 83 randomly
adjacent teeth, mandibular canal displacement, and the impac-
                                                                        selected cases by 3 methods: 1. panoramic radiograph only, 2.
tion of teeth were seen more frequently in ameloblastoma than
                                                                        CT only, 3. panoramic radiograph and CT simultaneously.
in odontogenic keratocyst, and cortical expansion showed
                                                                        The diagnoses were classified on 5-grade semiquantitative
more severely in ameloblastoma.
                                                                                 =                                  =
                                                                        scales: 1=must be odontogenic keratocyst; 2=may be odonto-
   The reliable interpretation of dental radiographs may be
                                                                                            =
                                                                        genic keratocyst; 3= odontogenic keratocyst or amelobla-
affected by a variety of biases, including the education, train-
                                                                                 =                         =
                                                                        stoma; 4=may be ameloblastoma; 5=must be ameloblastoma.
ing, and experience of the observer,16 which makes the obser-
                                                                        All observers were blinded to their own results and those of
ver difference in radiologic diagnosis. Choi et al.17 examined
                                                                        the other observers. After over 2 weeks, the observers dia-
the accuracy and inter- and intraobserver agreement in the
                                                                        gnosed them by same methods.
radiologic diagnosis of ameloblastoma and odontogenic
keratocyst on panoramic radiograph. They reported that                      3) Statistical analysis
experienced observer could differentiate more accurately than             Receiver operating characteristic (ROC) curves for each
inexperienced observer, and that the diagnostic accuracy                observers and groups were analysized and areas under the
highly correlated to the intraobserver agreement.                       curve were calculated. Intra- and interobserver agreement was
   But we didn’t find the studies about the difference of effec-        determined by using the kappa statistics with 95% confidence
tiveness by imaging modalities and observers in the differen-           intervals for each pairs of observers and groups. All statistical
tial diagnosis between ameloblastoma and odontogenic                    analysis were performed by the statistical software package

                                                                ─ 178 ─
Tae-In Gang, Kyung-Hoe Huh, Won-Jin Yi, Min-Suk Heo, Sam-Sun Lee, Jeong-Hwa Kim, Je-Woon Moon, Soon-Chul Choi


SPSS, release 10.0. (SPSS Inc., Chicago, USA).                                   (0.751), followed with panoramic radiograph only (0.748),
                                                                                 and was the lowest with CT only (0.672) (Fig. 4). When
                                                                                 observers diagnosed using panoramic radiograph only, the
                                   Results
                                                                                 areas under the curve were 0.765 for group 1, 0.763 for group
          1. ROC analysis                                                        2, and 0.748 for group 3. When observers interpreted using
                                                                                 CT only, the areas under the curve were 0.812 for group 1,
  The overall areas under the curve were the highest with
                                                                                 0.801 for group 2, and 0.672 for group 3. When observers
panoramic radiograph and CT (0.825), followed with CT only
                                                                                 examined using panoramic radiograph and CT simultane-
(0.758), and was the lowest with panoramic radiograph only
                                                                                 ously, the areas under the curve were 0.870 for group 1 and 2,
(0.757) (Fig. 1). The ROC curve areas of group 1 and 2 were
                                                                                 and 0.751 for group 3 (Fig. 5).
the highest with panoramic radiograph and CT (0.870, both),
followed with CT only (0.812 and 0.801, respectively), and
                                                                                               2. Intraobserver agreement
was the lowest with panoramic radiograph only (0.765 and
0.763, respectively) (Figs. 2, 3). The ROC curve areas of                          We found the various kappa values of 0.27 to 0.76 for the
group 3 were the highest with panoramic radiograph and CT                        intraobserver agreement. The kappa values varied between


                1                                                                                  1




              0.75                                                                               0.75
                                                                                 Sensitivity
Sensitivity




               0.5                                                                                0.5




              0.25                                                                               0.25
                                   Panoramic radiograph; area=0.757                                                  Panoramic radiograph; area=0.763
                                   CT; area=0.758                                                                    CT; area=0.801
                                   Panoramic radiograph and CT; area=0.825                                           Panoramic radiograph and CT; area=0.870
                                   Reference                                                                         Reference
                0                                                                                  0
                 0          0.25           0.5           0.75                1                          0     0.25           0.5           0.75                1
                                      1-Specificity                                                                      1-Specificity

Fig. 1. ROC curve of all observers.                                              Fig. 3. ROC curve of group 2.



                1                                                                                   1




              0.75                                                                               0.75
Sensitivity




                                                                                  Sensitivity




               0.5                                                                                0.5




              0.25                                                                               0.25
                                   Panoramic radiograph; area=0.765                                                  Panoramic radiograph; area=0.748
                                   CT; area=0.812                                                                    CT; area=0.672
                                   Panoramic radiograph and CT; area=0.870                                           Panoramic radiograph and CT; area=0.751
                                   Reference                                                                         Reference
                0                                                                                   0
                     0      0.25           0.5           0.75                1                       0        0.25           0.5            0.75               1
                                       1-Specificity                                                                     1-Specificity

Fig. 2. ROC curve of group 1.                                                    Fig. 4. ROC curve of group 3.

                                                                         ─ 179 ─
Diagnostic ability of differential diagnosis in ameloblastoma and odontogenic keratocyst by imaging modalities and observers


0.90
                                                                                                         Discussion
0.80
0.70                                                                               Frame and Wake18 reviewed the value of CT as an aid to
0.60                                                                            diagnosis and treatment planning in selected conditions affec-
0.50                                                                            ting the jaws and related structures, and many features of CT
0.40                                                                            which are helpful in diagnosis were demonstrated: 1. It is the
0.30                                                                            technique that provides an axial view of the tissue. In addi-
0.20                                                                            tion, coronal scans of the jaw can also be obtained to give
0.10                                                                            greater clarity; 2. A good display of the soft tissue is obtained,
0.00                                                                            both the normal anatomy such as muscles, and pathologic
       Panoramic radiograph                CT           Panoramic radiograph
                                                              and CT            lesions where extension into the adjacent tissues can be seen;
                    Group 1            Group 2               Group 3
                                                                                3. An assessment of tissue density is possible by determining
                                                                                the absorption values of the lesions. This indicates the possi-
Fig. 5. Areas under curve of individual groups.                                 ble make-up or contents of a structure or tumor as to whether
                                                                                it is predominantly fluid, cellular, or vascular; 4. A clearer
                                                                                picture is produced than with conventional radiographs, where
Table 1. Intraobserver agreement (kappa values)                                 there is often blurring because of superimposition of struc-
                              Panoramic                       Panoramic         tures outside the plane of interest. Mackenzie et al.19 reported
Group       Observer                            CT
                              radiograph                  radiograph and CT
                                                                                limitations of CT, which were possibility of failure of coronal
                A               0.61            0.49             0.76
   1                                                                            CT and streak artifacts on the scans by high density objects.
                B               0.59            0.56             0.72
                C               0.47            0.62             0.57           Cohen et al.20 reported that the soft tissue mass, destruction of
   2
                D               0.50            0.44             0.67           cortical bone, and extension of tumor into adjacent structures
                E               0.39            0.27             0.48           of mandibular ameloblastoma were clearly visualized using
   3
                F               0.32            0.52             0.47           CT. Abrahams and Oliverio21 reported that dental CT could
                                                                                provide a better means of assessing odontogenic cysts than
                                                                                conventional techniques (orthopantomographic, intraoral, and
Table 2. Interobserver agreement (kappa values)
                                                                                mandibular films), which are of limited usefulness because of
                        Panoramic                      Panoramic radiograph
   Group
                        radiograph
                                            CT
                                                             and CT             the curved configuration of the mandible. Kawai et al.22 made
                                                                                a comparative evaluation of the conventional radiography,
Group 1 and 2                 0.42          0.49               0.64
Group 2 and 3                 0.38          0.43               0.42             CT, magnetic resonance (MR) imaging of maxillary amelobla-
Group 1 and 3                 0.34          0.37               0.50             stoma, and reported that CT and MR imaging were superior to
                                                                                conventional radiography, that CT may be superior to MR
                                                                                imaging in the lesions bound by outlining bony tissues, but
0.49 and 0.76 for group 1, between 0.44 and 0.67 for group 2,
                                                                                that MR imaging was the best way of imaging in the recurrent
and between 0.27 and 0.52 for group 3. The kappa values
                                                                                lesions, which might not always be outlined by bony tissues.
varied between 0.32 and 0.61 for interpreting panoramic
                                                                                Iko et al.23 proposed that CT was the most sensitive imaging
radiograph only, between 0.27 and 0.62 for interpreting CT
                                                                                technique for detecting ameloblastomas, and Kurabayashi24
only, and between 0.47 and 0.76 for interpreting panoramic
                                                                                emphasized that CT was useful in demonstrating the extent of
radiograph and CT (Table 1).
                                                                                maxillofacial mass lesions in children and for surgical treat-
                                                                                ment planning. So the many studies about the utility of CT in
  3. Interobserver agreement
                                                                                diagnosis and treatment planning of ameloblastoma and
  Interobserver kappa values ranged from 0.34 to 0.64. The                      odontogenic keratocyst have been reported. But the difference
kappa values varied between 0.34 and 0.42 for using pano-                       of effectiveness by imaging modalities and observers in the
ramic radiograph only, between 0.37 and 0.49 for using CT                       differential diagnosis between ameloblastoma and odonto-
only, and between 0.42 and 0.64 for using panoramic radio-                      genic keratocyst does not appear to have been discussed suf-
graph and CT (Table 2).                                                         ficiently. We examined the diagnostic ability of each obser-
                                                                                vers and groups by image modalities. Diagnostic ability is

                                                                           ─ 180 ─
Tae-In Gang, Kyung-Hoe Huh, Won-Jin Yi, Min-Suk Heo, Sam-Sun Lee, Jeong-Hwa Kim, Je-Woon Moon, Soon-Chul Choi


measured by the area under the ROC curve. The closer the          good intraobserver agreement. The overall kappa values were
curve follows the left-hand border and then the top border of     fair-to-good interobserver agreement. The kappa values
the ROC space, the more accurate the test. The closer the         between group 1 and 2 were moderate-to-good interobserver
curve comes to the 45-degree diagonal of the ROC space, the       agreement, but the other kappa values were fair-to-moderate
less accurate the test.25 In this study, the ROC curve areas      interobserver agreement. The kappa values of using pano-
except for group 3 were the highest with interpretation using     ramic radiograph only and using CT only were fair-to-
panoramic radiograph and CT, followed by interpretation           moderate interobserver agreement, and those of using pano-
using CT only, and was the lowest with interpretation using       ramic radiograph and CT were moderate-to-good interobse-
panoramic radiograph only. As a result, in differential diagno-   rver agreement. As a general, when the observers who had
sis between ameloblastoma and odontogenic keratocyst the          experienced interpretation of CT over years diagnosed by
diagnostic ability of CT was superior to that of panoramic        using panoramic radiograph and CT simultaneously, it was a
radiograph, and combination of panoramic radiograph and CT        tendency to show higher intra- and interobserver agreement.
could increase the diagnostic ability more.                         Conclusionally, when making a differential diagnosis
   The overall difference was not found in diagnostic ability     between ameloblastoma and odontogenic keratocyst, com-
among groups in using panoramic radiograph only, but there        bination of panoramic radiograph and CT increases the
was difference in diagnostic ability of group 1 and 2 vs 3 in     diagnostic ability and the intra- and interobserver agreement,
using CT only, and combination panoramic radiograph and           especially in the experienced oral and maxillofacial radio-
CT. The group 1 and 2 were composed of the experienced            logists.
observers of CT, but group 3 was composed of the observers
who had not experienced with interpretation of CT. Although
                                                                                               References
the observers of group 3 had studied the characteristics of CT
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                                                                      stoma. J Oral Maxillof Surg 1984; 42 : 161-6.
chance, and negative values show worse than chance agree-
                                                                  10. Tanimoto K, Fujita M, Wada T, Koseki T, Fujiwara M, Uemura S.
ment. The following qualitative terms are used to interpret           Radiographic features of odonotogenic keratocyst in the mandibular
kappa statistics: poor, 0.00-0.20; fair, 0.21-0.40; moderate,         ramus - for the differential diagnosis from ameloblastoma -. Dent
0.41-0.60; good, 0.61-0.80; and perfect, 0.81-1.00.26 In this         Radiol 1982; 21 : 237-45.
                                                                  11. Choi KS. A radiographic study of differential diagnosis between
study the overall kappa values were fair-to-good intraobserver
                                                                      odontogenic keratocyst and unicystic ameloblastoma. Korean J Oral
agreement within a group. The kappa values of group 1 and 2           Maxillofac Radiol 1995; 25 : 17-25.
were moderate-to-good intraobserver agreement, but those of       12. Minami M, Kaneda T, Ozawa K, Yamamoto H, Itai Y, Ozawa M, et
group 3 were fair-to-moderate intraobserver agreement. The            al. Cystic lesions of the maxillomandibular region: MR imaging dis-
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                                                                      cysts. AJR Am J Roentgenol 1996; 166 : 943-9.
CT only were fair-to-good intraobserver agreement, but those      13. Jeong HG, Lee JY, Kim KD, Park CS. Clinicoradiologic differential
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    Oral Maxillofac Radiol 2000; 30 : 249-54.                             21. Abrahams JJ, Oliverio PJ. Odontogenic cysts: improved imaging with
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15. Soh BC, Heo MS, An CH, Choi M, Lee SS, Choi SC, et al. Radio-             H. Diagnostic imaging in two cases of recurrent maxillary amelobla-
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                                                                    ─ 182 ─

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Diagnostic ability of differential diagnosis in ameloblastoma and odontogenic keratocyst by imaging modalities and observers

  • 1. Korean Journal of Oral and Maxillofacial Radiology 2006; 36 : 177-82 Diagnostic ability of differential diagnosis in ameloblastoma and odontogenic keratocyst by imaging modalities and observers Tae-In Gang, Kyung-Hoe Huh, Won-Jin Yi**, Min-Suk Heo**, Sam-Sun Lee*, Jeong-Hwa Kim, Je-Woon Moon, Soon-Chul Choi* Department of Oral and Maxillofacial Radiology *Department of Oral and Maxillofacial Radiology, Dental Research Institute **Department of Oral and Maxillofacial Radiology, Dental Research Institute, and BK21 School of Dentistry, Seoul National University ABSTRACT Purpose : To evaluate the diagnostic ability in differentiating between ameloblastoma and odontogenic keratocyst according to the imaging modalities and observers. Materials and Methods : We evaluated thirty-six cases of ameloblastomas and forty-seven cases of odontogenic keratocysts all histologically confirmed. Six oral and maxillofacial radiologists diagnosed the lesions by 3 methods: using panoramic radiograph, using computed tomograph (CT), and using panoramic radiograph and CT. The observers were classified by 3 groups: group 1 had experienced over 10 years in oral and maxillofacial radiologic field, group 2 had experienced for 3-4 years, and group 3 was in the process of residentship. After over 2 weeks, the observers diagnosed them by the same methods. Results : The ROC curve areas except for group 3 were the highest with interpretation using panoramic radiograph and CT, followed by interpretation using CT only, and the lowest with interpretation using panoramic radiograph only. The overall difference was not found in diagnostic ability among groups in using panoramic radiograph only, but there was difference in diagnostic ability of group 1 and 2 vs 3 in using CT only, and combination panoramic radiograph and CT. Conclusions : To differentiate between ameloblastoma and odontogenic keratocyst more accurately, the experienced oral and maxillofacial radiologist should diagnose with combination of panoramic radiograph and CT. (Korean J Oral Maxillofac Radiol 2006; 36 : 177-82) KEY WORDS : Diagnostic Ability; Ameloblastoma; Odontogenic Keratocyst lingual cortical expansion is frequently present. Root resorp- Introduction tion of adjacent teeth to the tumor is common.1 Ameloblastomas are tumors of odontogenic epithelial Odontogenic keratocysts arise from cell rests of the dental origin. Theoretically, they may arise from cell rests of the lamina and make up 10 to 12 percent of all developmental enamel organ, from a developing enamel organ, from the odontogenic cysts.1 Histopathologically, they typically show a epithelial lining of an odontogenic cyst, or from the basal cells thin, friable wall, which is often difficult to enucleate from the of the oral mucosa.1 They make up 1% of all tumors of the bone in one piece, and have small satellite cysts within fibrous jaws,2 and 11% of odontogenic tumors.3 Although ameloblas- wall. Therefore odontogenic keratocysts often tend to recur tomas are histopathologically benign, they have been charac- after treatment.7 Radiographically odontogenic keratocysts terized as a tumor with marked tendency to recur.2,4 Radio- demonstrate a well-defined unilocular or multilocular radiolu- graphically ameloblastomas show an expansile unilocular or cency with smooth and often corticated margins. 1 Odonto- multilocular lesion with a “honeycomb or soap bubble” genic keratocysts tend to grow in and anteroposterior direction appearance and a sharp scalloping border. 5,6 Buccal and within the medullary cavity of the bone without causing obvi- ous bone expansion.1 Displacement of teeth adjacent to the Received October 4, 2006; accepted November 6, 2006 Correspondence to : Prof. Soon-Chul Choi cyst occurs more frequently than resorption.6 Department of Oral and Maxillofacial Radiology School of Dentistry, Seoul The surgical treatment of ameloblastomas is marginal or National University 28, Yeongeon-dong, Jongno-gu, Seoul, 110-749, Korea Tel) 82-2-2072-2622, Fax) 82-2-744-3919, E-mail) raychoi@snu.ac.kr block resection, but most odontogenic keratocysts are treated ─ 177 ─
  • 2. Diagnostic ability of differential diagnosis in ameloblastoma and odontogenic keratocyst by imaging modalities and observers by enucleation and curretage.8,9 The preoperative differential keratocyst. The purpose of the present study is to assess the diagnosis between ameloblastoma and odontogenic keratocyst diagnostic ability, intra- and interobserver agreement on is important to establish appropriate treatment planning, but radiologic differential diagnosis in ameloblastoma and ameloblastoma and odontogenic keratocyst are not much odontogenic keratocyst by imaging modalities and observers. different in development age or site, and sometimes they are radiologically similiar, which makes it not easy to differenti- Materials and Methods ate between them. The studies of the preoperative differential diagnosis 1. Materials between ameloblastoma and odontogenic keratocyst have We examined histopathologically confirmed cases, taken been reported over years. Tanimoto et al.10 reported that buc- preoperative panoramic radiograph and CT (IQ scanner, colingual expansion of the ramus caused by odontogenic Picker International, USA), from January 1997 to July 2003 in keratocysts was smaller than that caused by ameloblastomas. Seoul National University Dental Hospital. Thirty-six cases of Choi11 reported that lesional outline, the root resorption of ameloblastomas and forty-seven cases of odontogenic kerato- adjacent teeth, tooth displacement, and the loss of lamina dura cysts were selected. The cases showing the typical multilo- could be the parameters to differentiate odontogenic kerato- cular characteristics of the ameloblastoma and recurrent cases cyst and unilocular ameloblastoma. Minami et al.12 reviewed were not included. the magnetic resonance findings in patients with odontogenic keratocysts or ameloblastomas, and proposed that from the 2. Methods magnetic resonance (MR) findings of the walls, solid com- ponents, and the fluid contents, odontogenic keratocysts could 1) Observers be differentiated from ameloblastomas. Jeong13 et al. describ- The six observers who participated in the study were ed that maxillary lesions were more common in odontogenic divided three groups. Group 1 was composed of the two keratocyst, multilocular lesions were seen more frequently in observers (observer A, B), having experience over 10 years in ameloblastoma, and the external root resorption of adjacent oral and maxillofacial radiologic field. Group 2 was com- teeth showed more frequently in ameloblastoma. Tozaki et posed of the two observers (observer C, D), having experience al.14 reported that a rapidly enhancing area was detected in 4-5 years in oral and maxillofacial radiologic field. Group 3 cases of ameloblastoma, while no apparent rapid enhancement was composed of the two observers (observer E, F), in the was seen in the other cystic lesions, including odontogenic process of residentship. keratocyst on dynamic multislice helical computed tomograph 2) Diagnostic method (CT). Soh et al.15 described that the root resorption of the Each one of the six observers diagnosed the 83 randomly adjacent teeth, mandibular canal displacement, and the impac- selected cases by 3 methods: 1. panoramic radiograph only, 2. tion of teeth were seen more frequently in ameloblastoma than CT only, 3. panoramic radiograph and CT simultaneously. in odontogenic keratocyst, and cortical expansion showed The diagnoses were classified on 5-grade semiquantitative more severely in ameloblastoma. = = scales: 1=must be odontogenic keratocyst; 2=may be odonto- The reliable interpretation of dental radiographs may be = genic keratocyst; 3= odontogenic keratocyst or amelobla- affected by a variety of biases, including the education, train- = = stoma; 4=may be ameloblastoma; 5=must be ameloblastoma. ing, and experience of the observer,16 which makes the obser- All observers were blinded to their own results and those of ver difference in radiologic diagnosis. Choi et al.17 examined the other observers. After over 2 weeks, the observers dia- the accuracy and inter- and intraobserver agreement in the gnosed them by same methods. radiologic diagnosis of ameloblastoma and odontogenic keratocyst on panoramic radiograph. They reported that 3) Statistical analysis experienced observer could differentiate more accurately than Receiver operating characteristic (ROC) curves for each inexperienced observer, and that the diagnostic accuracy observers and groups were analysized and areas under the highly correlated to the intraobserver agreement. curve were calculated. Intra- and interobserver agreement was But we didn’t find the studies about the difference of effec- determined by using the kappa statistics with 95% confidence tiveness by imaging modalities and observers in the differen- intervals for each pairs of observers and groups. All statistical tial diagnosis between ameloblastoma and odontogenic analysis were performed by the statistical software package ─ 178 ─
  • 3. Tae-In Gang, Kyung-Hoe Huh, Won-Jin Yi, Min-Suk Heo, Sam-Sun Lee, Jeong-Hwa Kim, Je-Woon Moon, Soon-Chul Choi SPSS, release 10.0. (SPSS Inc., Chicago, USA). (0.751), followed with panoramic radiograph only (0.748), and was the lowest with CT only (0.672) (Fig. 4). When observers diagnosed using panoramic radiograph only, the Results areas under the curve were 0.765 for group 1, 0.763 for group 1. ROC analysis 2, and 0.748 for group 3. When observers interpreted using CT only, the areas under the curve were 0.812 for group 1, The overall areas under the curve were the highest with 0.801 for group 2, and 0.672 for group 3. When observers panoramic radiograph and CT (0.825), followed with CT only examined using panoramic radiograph and CT simultane- (0.758), and was the lowest with panoramic radiograph only ously, the areas under the curve were 0.870 for group 1 and 2, (0.757) (Fig. 1). The ROC curve areas of group 1 and 2 were and 0.751 for group 3 (Fig. 5). the highest with panoramic radiograph and CT (0.870, both), followed with CT only (0.812 and 0.801, respectively), and 2. Intraobserver agreement was the lowest with panoramic radiograph only (0.765 and 0.763, respectively) (Figs. 2, 3). The ROC curve areas of We found the various kappa values of 0.27 to 0.76 for the group 3 were the highest with panoramic radiograph and CT intraobserver agreement. The kappa values varied between 1 1 0.75 0.75 Sensitivity Sensitivity 0.5 0.5 0.25 0.25 Panoramic radiograph; area=0.757 Panoramic radiograph; area=0.763 CT; area=0.758 CT; area=0.801 Panoramic radiograph and CT; area=0.825 Panoramic radiograph and CT; area=0.870 Reference Reference 0 0 0 0.25 0.5 0.75 1 0 0.25 0.5 0.75 1 1-Specificity 1-Specificity Fig. 1. ROC curve of all observers. Fig. 3. ROC curve of group 2. 1 1 0.75 0.75 Sensitivity Sensitivity 0.5 0.5 0.25 0.25 Panoramic radiograph; area=0.765 Panoramic radiograph; area=0.748 CT; area=0.812 CT; area=0.672 Panoramic radiograph and CT; area=0.870 Panoramic radiograph and CT; area=0.751 Reference Reference 0 0 0 0.25 0.5 0.75 1 0 0.25 0.5 0.75 1 1-Specificity 1-Specificity Fig. 2. ROC curve of group 1. Fig. 4. ROC curve of group 3. ─ 179 ─
  • 4. Diagnostic ability of differential diagnosis in ameloblastoma and odontogenic keratocyst by imaging modalities and observers 0.90 Discussion 0.80 0.70 Frame and Wake18 reviewed the value of CT as an aid to 0.60 diagnosis and treatment planning in selected conditions affec- 0.50 ting the jaws and related structures, and many features of CT 0.40 which are helpful in diagnosis were demonstrated: 1. It is the 0.30 technique that provides an axial view of the tissue. In addi- 0.20 tion, coronal scans of the jaw can also be obtained to give 0.10 greater clarity; 2. A good display of the soft tissue is obtained, 0.00 both the normal anatomy such as muscles, and pathologic Panoramic radiograph CT Panoramic radiograph and CT lesions where extension into the adjacent tissues can be seen; Group 1 Group 2 Group 3 3. An assessment of tissue density is possible by determining the absorption values of the lesions. This indicates the possi- Fig. 5. Areas under curve of individual groups. ble make-up or contents of a structure or tumor as to whether it is predominantly fluid, cellular, or vascular; 4. A clearer picture is produced than with conventional radiographs, where Table 1. Intraobserver agreement (kappa values) there is often blurring because of superimposition of struc- Panoramic Panoramic tures outside the plane of interest. Mackenzie et al.19 reported Group Observer CT radiograph radiograph and CT limitations of CT, which were possibility of failure of coronal A 0.61 0.49 0.76 1 CT and streak artifacts on the scans by high density objects. B 0.59 0.56 0.72 C 0.47 0.62 0.57 Cohen et al.20 reported that the soft tissue mass, destruction of 2 D 0.50 0.44 0.67 cortical bone, and extension of tumor into adjacent structures E 0.39 0.27 0.48 of mandibular ameloblastoma were clearly visualized using 3 F 0.32 0.52 0.47 CT. Abrahams and Oliverio21 reported that dental CT could provide a better means of assessing odontogenic cysts than conventional techniques (orthopantomographic, intraoral, and Table 2. Interobserver agreement (kappa values) mandibular films), which are of limited usefulness because of Panoramic Panoramic radiograph Group radiograph CT and CT the curved configuration of the mandible. Kawai et al.22 made a comparative evaluation of the conventional radiography, Group 1 and 2 0.42 0.49 0.64 Group 2 and 3 0.38 0.43 0.42 CT, magnetic resonance (MR) imaging of maxillary amelobla- Group 1 and 3 0.34 0.37 0.50 stoma, and reported that CT and MR imaging were superior to conventional radiography, that CT may be superior to MR imaging in the lesions bound by outlining bony tissues, but 0.49 and 0.76 for group 1, between 0.44 and 0.67 for group 2, that MR imaging was the best way of imaging in the recurrent and between 0.27 and 0.52 for group 3. The kappa values lesions, which might not always be outlined by bony tissues. varied between 0.32 and 0.61 for interpreting panoramic Iko et al.23 proposed that CT was the most sensitive imaging radiograph only, between 0.27 and 0.62 for interpreting CT technique for detecting ameloblastomas, and Kurabayashi24 only, and between 0.47 and 0.76 for interpreting panoramic emphasized that CT was useful in demonstrating the extent of radiograph and CT (Table 1). maxillofacial mass lesions in children and for surgical treat- ment planning. So the many studies about the utility of CT in 3. Interobserver agreement diagnosis and treatment planning of ameloblastoma and Interobserver kappa values ranged from 0.34 to 0.64. The odontogenic keratocyst have been reported. But the difference kappa values varied between 0.34 and 0.42 for using pano- of effectiveness by imaging modalities and observers in the ramic radiograph only, between 0.37 and 0.49 for using CT differential diagnosis between ameloblastoma and odonto- only, and between 0.42 and 0.64 for using panoramic radio- genic keratocyst does not appear to have been discussed suf- graph and CT (Table 2). ficiently. We examined the diagnostic ability of each obser- vers and groups by image modalities. Diagnostic ability is ─ 180 ─
  • 5. Tae-In Gang, Kyung-Hoe Huh, Won-Jin Yi, Min-Suk Heo, Sam-Sun Lee, Jeong-Hwa Kim, Je-Woon Moon, Soon-Chul Choi measured by the area under the ROC curve. The closer the good intraobserver agreement. The overall kappa values were curve follows the left-hand border and then the top border of fair-to-good interobserver agreement. The kappa values the ROC space, the more accurate the test. The closer the between group 1 and 2 were moderate-to-good interobserver curve comes to the 45-degree diagonal of the ROC space, the agreement, but the other kappa values were fair-to-moderate less accurate the test.25 In this study, the ROC curve areas interobserver agreement. The kappa values of using pano- except for group 3 were the highest with interpretation using ramic radiograph only and using CT only were fair-to- panoramic radiograph and CT, followed by interpretation moderate interobserver agreement, and those of using pano- using CT only, and was the lowest with interpretation using ramic radiograph and CT were moderate-to-good interobse- panoramic radiograph only. As a result, in differential diagno- rver agreement. As a general, when the observers who had sis between ameloblastoma and odontogenic keratocyst the experienced interpretation of CT over years diagnosed by diagnostic ability of CT was superior to that of panoramic using panoramic radiograph and CT simultaneously, it was a radiograph, and combination of panoramic radiograph and CT tendency to show higher intra- and interobserver agreement. could increase the diagnostic ability more. Conclusionally, when making a differential diagnosis The overall difference was not found in diagnostic ability between ameloblastoma and odontogenic keratocyst, com- among groups in using panoramic radiograph only, but there bination of panoramic radiograph and CT increases the was difference in diagnostic ability of group 1 and 2 vs 3 in diagnostic ability and the intra- and interobserver agreement, using CT only, and combination panoramic radiograph and especially in the experienced oral and maxillofacial radio- CT. The group 1 and 2 were composed of the experienced logists. observers of CT, but group 3 was composed of the observers who had not experienced with interpretation of CT. Although References the observers of group 3 had studied the characteristics of CT 1. Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and maxillo- of ameloblastoma and odontogenic keratocyst by textbook facial pathology. W.B. Saunders Co.; 1995. p. 496-512. before this study, most cases of this study were not shown the 2. Small IA, Waldron CA. Ameloblastomas of the jaws. Oral Surg Oral typical characteristics of them. In addition, they did not Med Oral Pathol 1955; 8 : 281-97. analyse them accurately even in some cases showing typical 3. Regezi JA. Odontogenic tumor: Analysis of 706 cases. J Oral Surg 1978; 36 : 771-8. characteristics. We could analogize that the experience of 4. Williams TP. Management of ameloblastoma: A changing perspec- radiologic diagnosis was an important factor for differential tive. J Oral Maxillofac Surg 1993; 51: 1064-70. diagnosis between ameloblastoma and odontogenic kerato- 5. Hertzanu Y, Mendelsohr DB, Cohen MA. Computed tomography of cyst, and the result of this study corresponded to that of mandibular ameloblastoma. J Comput Assist Tomogr 1984; 8 : 220-3. 6. Brannon RB. The odontogenic keratocyst: A clinicopathologic study previous study, which the observers, having the experience of of 312 cases, Part I. Clinical features. Oral Surg Oral Med Oral Pathol interpretation over 3 years could differentiate more accurately 1976; 42 : 54-72. than those of below 3 years.17 7. Brannon RB. The odontogenic keratocyst: A clinicopathologic study Kappa is defined as the difference between observed and of 312 cases, Part II. Histologic features. Oral Surg Oral Med Oral Pathol 1977; 43 : 233-55. expected agreement expressed as a fraction of the maximum 8. Shafer WG, Hine MK, Levy BM. Textbook of oral pathology. 4th ed. difference. The kappa value is 1 when agreement is perfect, W.B. Saunders Co.; 1983. p. 276-85. but a kappa value of 0 indicates agreement is only due to 9. Gardner DG. A pathologist’s approach to the treatment of amelobla- stoma. J Oral Maxillof Surg 1984; 42 : 161-6. chance, and negative values show worse than chance agree- 10. Tanimoto K, Fujita M, Wada T, Koseki T, Fujiwara M, Uemura S. ment. The following qualitative terms are used to interpret Radiographic features of odonotogenic keratocyst in the mandibular kappa statistics: poor, 0.00-0.20; fair, 0.21-0.40; moderate, ramus - for the differential diagnosis from ameloblastoma -. Dent 0.41-0.60; good, 0.61-0.80; and perfect, 0.81-1.00.26 In this Radiol 1982; 21 : 237-45. 11. Choi KS. A radiographic study of differential diagnosis between study the overall kappa values were fair-to-good intraobserver odontogenic keratocyst and unicystic ameloblastoma. Korean J Oral agreement within a group. The kappa values of group 1 and 2 Maxillofac Radiol 1995; 25 : 17-25. were moderate-to-good intraobserver agreement, but those of 12. Minami M, Kaneda T, Ozawa K, Yamamoto H, Itai Y, Ozawa M, et group 3 were fair-to-moderate intraobserver agreement. The al. 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