Application of machine learning in the preoperative radiomic diagnosis of ameloblastoma and odontogenic keratocyst based on cone-beam CT.

OBJECTIVES: Preoperative diagnosis of oral ameloblastoma (AME) and odontogenic keratocyst (OKC) has been a challenge in dentistry. This study uses radiomics approaches and machine learning (ML) algorithms to characterize cone-beam CT (CBCT) image features for the preoperative differential diagnosis of AME and OKC and compares ML algorithms to expert radiologists to validate performance. METHODS: We retrospectively collected the data of 326 patients with AME and OKC, where all diagnoses were confirmed by histopathologic tests. A total of 348 features were selected to train six ML models for differential diagnosis by a 5-fold cross-validation. We then compared the performance of ML-based diagnoses to those of radiologists. RESULTS: Among the six ML models, XGBoost was effective in distinguishing AME and OKC in CBCT images, with its classification performance outperforming the other models. The mean precision, recall, accuracy, F1-score, and area under the curve (AUC) were 0.900, 0.807, 0.843, 0.841, and 0.872, respectively. Compared to the diagnostics by radiologists, ML-based radiomic diagnostics performed better. CONCLUSIONS: Radiomic-based ML algorithms allow CBCT images of AME and OKC to be distinguished accurately, facilitating the preoperative differential diagnosis of AME and OKC. ADVANCES IN KNOWLEDGE: ML and radiomic approaches with high-resolution CBCT images provide new insights into the differential diagnosis of AME and OKC.

Ossifying fibroma mimiking jaw tumour: A radiographic dilema.

Fibro-osseous lesions (FOLs) of the craniomaxillofacial region comprise a group of developmental, dysplastic, and neoplastic alterations. FOLs include ossifying fibromas (OF), cemento-ossifying fibroma (COF), familial gigantiform cementoma (FGC), fibrous dysplasia (FD), and cemento-osseous dysplasia (COD). Evidence suggests that some FOL, especially FD and OF may have a risk of spontaneous malignant transformation. This report documents a rare case of malignant transformation of ossifying fibromas of the jaw and the probable cause for same. Although it is rare, the clinician should have a complete follow up to observe such changes among the patients having FOLs.

Evaluation of deep learning for detecting intraosseous jaw lesions in cone beam computed tomography volumes.

OBJECTIVE: The study aim was to develop and assess the performance of a deep learning (DL) algorithm in the detection of radiolucent intraosseous jaw lesions in cone beam computed tomography (CBCT) volumes. STUDY DESIGN: A total of 290 CBCT volumes from more than 12 different scanners were acquired. Fields of view ranged from 6 × 6 × 6 cm to 18 × 18 × 16 cm. CBCT volumes contained either zero or at least one biopsy-confirmed intraosseous lesion. 80 volumes with no intraosseous lesions were included as controls and were not annotated. 210 volumes with intraosseous lesions were manually annotated using ITK-Snap 3.8.0. 150 volumes (10 control, 140 positive) were presented to the DL software for training. Validation was performed using 60 volumes (30 control, 30 positive). Testing was performed using the remaining 80 volumes (40 control, 40 positive). RESULTS: The DL algorithm obtained an adjusted sensitivity by case, specificity by case, positive predictive value by case, and negative predictive value by case of 0.975, 0.825, 0.848, and 0.971, respectively. CONCLUSIONS: A DL algorithm showed moderate success at lesion detection in their correct locations, as well as recognition of lesion shape and extent. This study demonstrated the potential of DL methods for intraosseous lesion detection in CBCT volumes.

Radiolucent Lesions of the Jaws: An Attempted Demonstration of the Use of Co-Word Analysis to List Main Similar Pathologies.

(1) Background: Many radiolucent jaw lesions exist, and they often show a radiographic resemblance, rendering diagnosis a challenging act. Closely related lesions should be frequently mentioned together in the academic literature, which might be helpful for junior practitioners in determining their differential diagnosis. The usefulness of bibliometric analysis in this respect has yet to be demonstrated. (2) Methods: This study evaluated academic publications on radiolucent jaw lesions, as indexed by the Web of Science Core Collection database. The mentions of radiolucent jaw lesions were extracted from the complete bibliographic records of the publications, and co-word analyses were conducted with the aid of VOSviewer. (3) Results: Based on 1897 papers, visualization maps were synthesized to evaluate co-occurrences of the radiolucent jaw lesions. Ameloblastoma was frequently mentioned together with odontogenic keratocyst, dentigerous cyst, and radicular cyst. Osseous dysplasia was co-mentioned with osteomyelitis, ossifying fibroma, odontoma, fibrous dysplasia, and apical periodontitis. (4) Conclusions: The co-word analysis, a form of bibliometric analysis, could demonstrate a relatedness of radiolucent jaw lesions that could be considered at differential diagnosis.

Deep learning based diagnosis for cysts and tumors of jaw with massive healthy samples.

We aimed to develop an explainable and reliable method to diagnose cysts and tumors of the jaw with massive panoramic radiographs of healthy peoples based on deep learning, since collecting and labeling massive lesion samples are time-consuming, and existing deep learning-based methods lack explainability. Based on the collected 872 lesion samples and 10,000 healthy samples, a two-branch network was proposed for classifying the cysts and tumors of the jaw. The two-branch network is firstly pretrained on massive panoramic radiographs of healthy peoples, then is trained for classifying the sample categories and segmenting the lesion area. Totally, 200 healthy samples and 87 lesion samples were included in the testing stage. The average accuracy, precision, sensitivity, specificity, and F1 score of classification are 88.72%, 65.81%, 66.56%, 92.66%, and 66.14%, respectively. The average accuracy, precision, sensitivity, specificity, and F1 score of classification will reach 90.66%, 85.23%, 84.27%, 93.50%, and 84.74%, if only classifying the lesion samples and healthy samples. The proposed method showed encouraging performance in the diagnosis of cysts and tumors of the jaw. The classified categories and segmented lesion areas serve as the diagnostic basis for further diagnosis, which provides a reliable tool for diagnosing jaw tumors and cysts.

The importance of clinical and radiological findings for the definitive histopathologic diagnosis of benign fibro-osseous lesions of the jaws: Study of 276 cases.

PURPOSE: Benign fibro-osseous lesions (BFOLs) are heterogeneous intraosseous disease processes sharing overlapping histopathologic features and demonstrate a wide range of biological behavior. The aim of this study is to highlight the importance of clinical and radiological findings for the definitive histopathologic diagnosis of benign fibro-osseous lesions of the jaws. MATERIALS AND METHODS: This is a cross-sectional retrospective study evaluating the three main groups of BFOLs: Cemento-osseous dysplasia, ossifying fibromas, and fibrous dysplasia. Previously diagnosed BFOL cases were searched for thoroughly from the archives of Istanbul University, Institute of Oncology, Department of Tumor Pathology in between 2005 and 2015. Clinical and radiological data of these cases were identified and recorded. The histopathologic features were reevaluated from the H&E-stained slides. RESULTS: A total of 276 BFOL cases were identified and reevaluated for the study. The most common BFOL type was cemento-osseous dysplasia (n = 135), followed by cemento-ossifying fibroma (n = 77), fibrous dysplasia (n = 53) and juvenile ossifying fibroma (n = 11). The female/male ratio was 3.4:1 with a strong predilection for the 4th decade (n = 102). The mandible (n = 209) was the predominantly affected jaw. Prominent osteoblastic rimming around the bony lesion was commonly observed for cemento-ossifying fibroma (n = 60/77), followed by cemento-osseous dysplasia (n = 23/135). Predominant ossification patterns showed some differences among the groups. The most common radiological feature was the mixed radiolucent/radiopaque internal structure. CONCLUSION: Our results document various clinical, radiological and histopathologic findings of BFOLs. Even some histopathologic differences are observed, clinical and radiographic correlation is mandatory prior to the definitive histopathologic diagnosis of BFOLs for the prevention of possible misdiagnoses or inappropriate treatments.

Pathogenesis and characteristics of large ameloblastoma of the jaw: a report of two rare cases.

Ameloblastoma is a common odontogenic epithelial tumor that exhibits various biological behaviors, ranging from simple cystic expansion to aggressive solid masses characterized by local invasiveness, a high risk of recurrence, and even malignant transformation. We report on two cases of unusually large solid ameloblastomas. We detected epithelial-mesenchymal transition-related gene expression and HRAS gene single nucleotide polymorphisms, providing possible molecular evidence of mesenchymal morphological changes in ameloblastoma. The detailed analysis of the pathogenesis of these two cases of ameloblastoma may deepen our understanding of this rare disease and offer promising targets for future targeted therapy.

Computer tomographic differential diagnosis of ameloblastoma and odontogenic keratocyst: classification using a convolutional neural network.

OBJECTIVE: To analyse the automatic classification performance of a convolutional neural network (CNN), Google Inception v3, using tomographic images of odontogenic keratocysts (OKCs) and ameloblastomas (AMs). METHODS: For construction of the database, we selected axial multidetector CT images from patients with confirmed AM (n = 22) and OKC (n = 18) based on a conclusive histopathological report. The images (n = 350) were segmented manually and data augmentation algorithms were applied, totalling 2500 images. The k-fold × five cross-validation method (k = 2) was used to estimate the accuracy of the CNN model. RESULTS: The accuracy and standard deviation (%) of cross-validation for the five iterations performed were 90.16 ± 0.95, 91.37 ± 0.57, 91.62 ± 0.19, 92.48 ± 0.16 and 91.21 ± 0.87, respectively. A higher error rate was observed for the classification of AM images. CONCLUSION: This study demonstrated a high classification accuracy of Google Inception v3 for tomographic images of OKCs and AMs. However, AMs images presented the higher error rate.

18F-FDG PET/CT in the Hyperparathyroidism-Jaw-Tumor Syndrome.

ABSTRACT: Hyperparathyroidism-jaw-tumor syndrome (HPT-JTS) is a rare autosomal dominant disorder. A typical manifestation of HPT-JTS is the association of jaw-ossifying fibroma with primary hyperparathyroidism. Due to its rarity and diversity in its manifestations, it is a challenging diagnosis. A 33-year-old woman was referred due to painful swelling of the right maxilla suggestive of malignancy. The clinical presentations were not conclusive until she underwent F18-fluorodeoxyglucose positron emission tomography/computed tomography (F18-FDG PET/CT). F18-FDG PET/CT proved to be a useful tool to assist the clinicians in visualizing the "bigger picture" and, therefore all manifestation as pieces of "one puzzle."

Odontogenic keratocyst and ameloblastoma: radiographic evaluation.

OBJECTIVES: To describe the radiographic features of odontogenic keratocysts (OKCs) and ameloblastomas and to compare the radiographic findings between these 2 lesions. METHODS: Radiographs of OKCs and ameloblastomas were retrospectively reviewed. Location, border, shape, association with impacted tooth, tooth displacement, root resorption, and bone expansion were evaluated. Chi-squared or Fisher's exact tests were used for statistical analysis. A p value < 0.05 was considered to indicate statistical significance. RESULTS: One hundred OKCs and 101 ameloblastomas were reviewed. The ratios of maxilla to mandible were 1:1.4 and 1:9.1 in OKCs and ameloblastomas, respectively. All evaluated features significantly differed between OKCs and ameloblastomas (p ≤ 0.001). Most OKCs showed smooth border (60%) and unilocular shape (82%), while most ameloblastomas showed scalloped border (77.2%) and multilocular shape (68.3%). Association with impacted tooth was found in 47% of OKCs and 18.8% of ameloblastomas. Adjacent tooth displacement was found in 33.7% of OKCs and 55.8% of ameloblastomas. Root resorption was more common in ameloblastomas (66.7%) than in OKCs (7%). Bone expansion was also more common in ameloblastomas (96.3%) than in OKCs (63.6%). CONCLUSION: A unilocular radiolucent lesion with smooth border, no adjacent tooth displacement, no root resorption and causing mild or no bone expansion is suggestive of an OKC rather than an ameloblastoma.

Fibula osteal flap with proximal peroneal perforator skin paddle for composite oromandibular reconstruction: A case report.

RATIONALE: Cutaneous perforators of peroneal vessels are divided into proximal and distal perforators on the basis of perforator distributions and musculocutaneous or septocutaneous properties. The traditional fibular osteocutaneous free flap is raised over the distal two-thirds of the fibula with a skin paddle based on distal perforators, which is affixed to the posterior crural septum. However, the skin pedicle may not be available due to anatomic variations or intraoperative injuries. Herein, because of the absence of distal perforators, we reserved and expropriated proximal perforators originating from the musculocutaneous branch of the superior part of the peroneal artery before it divided into nutrient and arcuate arteries and successfully harvested a separate osteal fibula and proximal perforator skin paddle with a single vascular pedicle-peroneal vessel. PATIENT CONCERNS: A 62-year-old man with a 6-month history of mandibular swelling and soft tissue invasion was referred to us. DIAGNOSIS: Panoramic radiography and computed tomography showed an irregular radiolucent lesion of the mandibular body, and histopathological analysis confirmed a follicular-pattern ameloblastoma. INTERVENTIONS: The diseased mandible and soft tissue were resected and reconstructed with a vascularized fibular osteal flap with the proximal perforator skin paddle. OUTCOMES: The mandibular contour was successfully restored; the skin paddle in the mouth was in good condition after 8 months of follow-up. LESSONS: The proximal perforator is reliable and practical for supplying a skin paddle and has significant potential for future applications. We recommend reserving the proximal perforator skin paddle as a backup flap when planning to raise a fibula flap, since unavailability or injury of the traditional fibular skin island based on distal perforators occurs frequently. This approach can avoid the exploration for a second donor site, save surgical time, and reduce surgical complexity. Moreover, we anticipate more frequent use of the proximal perforator flap in the future because of its flexibility and large volume, and since it can be combined with the osteal fibula or fibular osteocutaneous flap. However, an understanding of the traits of the proximal perforator and determination of its peroneal origin by computed tomography angiography is crucial for predesigning fibular osteal flaps with a proximal perforator skin paddle.

Residual cyst of the jaws: A clinico-pathologic study of this seemingly inconspicuous lesion.

OBJECTIVES: Residual cysts are relatively rare inflammatory cysts of the jaws. They are essentially radicular cysts without the presence of the offending dentition. These lesions have the ability to destroy bone within the jaws without any symptoms. Moreover, they can mimic more aggressive cysts and tumours on radiographs. The aim of this study was to describe the clinico-pathological features of residual cysts in order to discern them from other cystic lesions as well as analyse their management and recurrence patterns. MATERIALS AND METHODS: Sixty-four histopathologically confirmed residual cysts were analysed based on their clinical, radiological and histopathological features. Their management and follow-up were also noted. RESULTS: The majority of lesions presented in elderly (46.8%), edentulous patients (60.9%) and were most commonly found in the posterior regions of the mandible (51.6%). Clinico-pathological features that aided in their diagnosis included long-standing history with slow growing swelling and presence of well-defined, unilocular cystic lesion associated with previously extracted dentition. Enucleation was a successful method in the management of residual cysts with very low recurrence rate (1.6%). Two patients (3.1%) developed squamous cell carcinoma from the cyst lining. CONCLUSION: Residual cysts should be high on the list of differential diagnosis when elderly, edentulous patients present with cystic lesions in the jaws compared to dentate patients (P<0.01). All lesions should by biopsied and sent for histopathological examination along with radiological correlation as they have the potential to transform into primary intra-osseous squamous cell carcinoma with devastating consequences to the patient.

Lung metastasis, an incidental finding in maxilla - A case report.

The aim of this article was to present an asymptomatic lesion with insignificant clinical findings which turned out to be metastatic lesion in the jaws with primary in lung. The most common site of lung metastasis in the orofacial region is the mandible, but in our case it was seen in the maxilla. Metastases to the jaw bones occur in later stages. Hence, a careful examination of patients with jaw bone lesions is strongly suggested. Metastasis to the jaw should be considered while doing oral examination as observed in the current case because such lesions usually develop at terminal stage of cancer.

Defining secure surgical bone margins in head and neck squamous cell carcinomas: The diagnostic impact of intraoperative cytological assessment of bone resection margins compared with preoperative imaging.

BACKGROUND: Imaging provides crucial staging information for treatment planning of head and neck squamous cell carcinomas (HNSCCs). Despite technical progress in imaging techniques, defining the extent of bone involvement preoperatively remains challenging and requires intraoperative information to control for adequate resection. The intraoperative cytological assessment of the bone resection margins (ICAB) provides information whether bone is infiltrated by carcinoma. The aim of this study was to assess the diagnostic value of preoperative imaging compared with ICAB in order to achieve carcinoma-free bone margins. MATERIALS AND METHODS: 108 HNSCC patients underwent preoperative computed tomography (CT), magnetic resonance imaging (MRI) and orthopantomogram (OPG) for staging and surgical planning. Curative resection was planned based on imaging. Intraoperatively, the resection margins were controlled by ICAB. The diagnostic value of preoperative imaging and ICAB was assessed with reference to the histological findings. RESULTS: CT showed a sensitivity of 89.7%, specificity of 63.0%, positive predictive value (PPV) of 85.9%, and negative predictive value (NPV) of 70.8%. MRI revealed a sensitivity of 45.5%, specificity of 66.7%, PPV of 71.4% and NPV of 40.0%. OPG-imaging had a sensitivity of 64.7%, specificity of 76.2%, PPV of 81.5%, NPV 57.1%. In comparison, ICAB provided a sensitivity of 78.6%, specificity of 95.7%, PPV 73.3%, and NPV 96.7%. The accuracy was 82.1%, 52.9%, 69.0%, and 93.5% for CT, MRI, OPG, and ICAB, respectively. CONCLUSION: Preoperative imaging lacks accuracy in defining adequate bone resection margins, compared with ICAB. ICAB supports preoperative imaging and intraoperative frozen sections to improve bone margin control.

Using CT texture analysis to differentiate cystic and cystic-appearing odontogenic lesions.

PURPOSE: Cystic and cystic-appearing odontogenic lesions of the jaw may appear similar on CT imaging. Accurate diagnosis is often difficult although the relationship of the lesion to the tooth root or crown may offer a clue to the etiology. The purpose of this study was to evaluate CT texture analysis as an aid in differentiating cystic and cystic-appearing odontogenic lesions of the jaw. METHODS: This was an IRB-approved retrospective study including 42 pathology-proven dentigerous cysts, 37 odontogenic keratocysts, and 19 ameloblastomas. Each lesion was manually segmented on axial CT images, and textural features were analyzed using an in-house-developed Matlab-based texture analysis program that extracted 47 texture features from each segmented volume. Statistical analysis was performed comparing all pairs of the three types of lesions. RESULTS: Pairwise analysis revealed that nine histogram features, one GLCM feature, three GLRL features, two Laws features, four GLGM features and two Chi-square features showed significant differences between dentigerous cysts and odontogenic keratocysts. Four histogram features and one Chi-square feature showed significant differences between odontogenic keratocysts and ameloblastomas. Two histogram features showed significant differences between dentigerous cysts and ameloblastomas. CONCLUSIONS: CT texture analysis may be useful as a noninvasive method to obtain additional quantitative information to differentiate cystic and cystic-appearing odontogenic lesions of the jaw.

MAID chemotherapy regimen as a treatment strategy for metastatic malignant ameloblastoma: A case report.

RATIONALE: Ameloblastoma is generally characterized as a benign tumor originating in odontogenic epithelium. However, few cases of metastatic malignant ameloblastoma have also been reported. Due to the low incidence of malignant ameloblastoma, there is no established treatment regimen. To explore effective treatment for malignant ameloblastoma, we reported this case study. PATIENTS CONCERNS: This report described a case of a 28-year-old malignant ameloblastoma female patient with multiple metastasis (brain and lung). DIAGNOSES: The patient presented ameloblastoma of the left mandible in 2012. Three years later, local recurrence and brain metastasis was observed during a follow-up examination. Five years later, malignant ameloblastoma was detected by imaging and immunohistochemistry in the bilateral multiple pulmonary nodules and mediastinal lymph nodes. INTERVENTIONS: The patient was initially treated with tumor resection. Three years later after local recurrence and brain metastasis, she was accepted the extensive mandibulectomy supplemented with brain stereotactic body radiotherapy (SBRT). When diagnosed with pulmonary metastasis, the patient received combined chemotherapy regimen of MAID (mesna, adriamycin, ifosfamide and dacarbazine) for 6 cycles. OUTCOMES: The efficacy evaluation was partial remission (PR) after the 6 cycles of MAID. The last patient follow-up was July 24th 2018, and no evidence of progression was observed. The progression-free survival (PFS) of the patient was more than 9 months. LESSONS: Surgical resection is the optimal treatment for locally recurrent ameloblastoma. SBRT may be an effective treatment for unresectable oligometastasis of malignant ameloblastoma. Finally, combined chemotherapy of MAID showed encouraging effects in the management of metastatic malignant ameloblastoma.

Oral malignant melanoma of alveolar ridge.

Malignancy of melanocytes, a pigment-producing cell, is referred as malignant melanoma (MM) which occur basically on skin and oral mucous membrane, but as well found in ears, eyes, gastrointestinal tract and genital mucosa. Oral melanomas has propensity to metastasise and invade more voluntarily than other malignant counterparts. Here we present a case of 52-year-old male patient with a chief symptom of blackening of gums in the upper front tooth region. In dental history, the patient revealed history of faulty artificial prosthesis fixed in the same region since 6 months. On the basis of a through clinical assessment, a provisional opinion of oral malignant melanoma, was prepared. On histopathological and immunohistochemical analysis with S-100 and homatropine methylbromide 45 the diagnosis of MM was confirmed.

Three-dimensional radiographic features of ameloblastoma and cystic lesions in the maxilla.

OBJECTIVES: To characterize the radiographic features of maxillary ameloblastoma (AM), odontogenic keratocyst (OKC) and dentigerous cyst (DC) comparatively by using spiral CT and cone beam CT (CBCT). METHODS: Clinical records, histopathological reports, and nonenhanced spiral CT or CBCT images of 191 consecutive patients with primary maxillary AMs, OKCs, or DCs were retrospectively acquired, and radiographic features were analyzed. RESULTS: The study included 118 males and 73 females (age: 5-84 years). 72.0% of AMs and 84.3% of OKCs originated from the posterior maxilla, while 69.6% of DCs occurred in the anterior maxilla. Among 25 AMs, 44.0% were of desmoplastic type, with honey-combed appearance. 84.0% of AMs were circular or oval in shape, 84.0% expanded buccally, and 36.0% invade the nasal floor. Among 89 OKCs of 88 patients, 61.8% were circular or oval, 58.4% expanded buccally, 49.4% were dentigerous, 41.6% nearly filled the maxillary sinus, and 13.5% invaded the nasal floor. 93.7% (74/79) of DCs enveloped a single tooth, and the tooth-cyst relationship was centripetal in 35, eccentric in 30, and circumferential in 9. Moreover, 98.2% (55/56) of the cysts enveloping a supernumerary tooth were DCs, while 80.9% (38/47) of the cysts enveloping the third molar were OKCs. CONCLUSIONS: Maxillary AMs tend to grow with buccal expansion and invade the nasal floor, and DAs with honey-combed lobularity are common. Maxillary OKCs have variant shapes and tend to invaginate the maxillary sinus. The tooth-cyst relationship of dentigerous OKCs and DCs can be centripetal, eccentric, or circumferential.