FRINK Linked Data Fragments server

Matches in SemOpenAlex for { <https://semopenalex.org/work/W4361002039> ?p ?o ?g. }

• W4361002039 endingPage "900" @default.
• W4361002039 startingPage "897" @default.
• W4361002039 abstract "Sclerosing odontogenic carcinoma (SOC) is very rare. Moreover, it was only formally adopted in the 4th edition of the World Health Organization (WHO) classification of head and neck tumours in 2017.1Odell E.W. Koutlas I. Sclerosing odontogenic carcinoma.in: El-Naggar A.K. Chan J.K.C. Grandis J.R. WHO Classification of Head and Neck Tumours. 4th ed. IARC, Lyon2017: 209-210Google Scholar SOC is a locally aggressive low grade malignancy primarily diagnosed based on its microscopic appearance (i.e., skeletal muscle invasion or perineural infiltration).1Odell E.W. Koutlas I. Sclerosing odontogenic carcinoma.in: El-Naggar A.K. Chan J.K.C. Grandis J.R. WHO Classification of Head and Neck Tumours. 4th ed. IARC, Lyon2017: 209-210Google Scholar However, the scarcity of reported cases (less than 15) precludes malignancy grading.2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar, 3Soluk-Tekkesin M. Wright J.M. The World Health Organization Classification of odontogenic lesions: a summary of the changes of the 2022 5th ed.Turk J Pathol. 2022; 38: 168-184Google Scholar, 4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar The consensus among modern pathologists is that SOC has no metastatic potential,2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar,3Soluk-Tekkesin M. Wright J.M. The World Health Organization Classification of odontogenic lesions: a summary of the changes of the 2022 5th ed.Turk J Pathol. 2022; 38: 168-184Google Scholar which was added to the SOC definition in the 5th edition of the WHO classification of head and neck tumours, available online in 2022.4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar Nevertheless, the true malignant nature of SOC remains controversial because reports on metastasis are lacking.1Odell E.W. Koutlas I. Sclerosing odontogenic carcinoma.in: El-Naggar A.K. Chan J.K.C. Grandis J.R. WHO Classification of Head and Neck Tumours. 4th ed. IARC, Lyon2017: 209-210Google Scholar, 2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar, 3Soluk-Tekkesin M. Wright J.M. The World Health Organization Classification of odontogenic lesions: a summary of the changes of the 2022 5th ed.Turk J Pathol. 2022; 38: 168-184Google Scholar, 4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar, 5Ide F. Ito Y. Muramatsu T. et al.Sclerosing odontogenic carcinoma: a morphologic pattern or pathologic entity?.Oral Surg Oral Med Oral Pathol Oral Radiol. 2013; 115: 839Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar Herein, we describe a case of SOC with solitary lymph node metastasis. Such biological behaviour has never been documented.4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar Therefore, this case provides undeniable evidence that SOC metastases are possible. A 41-year-old woman presented with diffuse swelling of the left mandible. Clinical examinations showed a hard buccal expansion of the body of the mandible. Furthermore, axial post-contrast computed tomography (CT) revealed a heterogeneously enhanced space-occupying mass in the left mandible with the destruction of the buccal and lingual cortices (Fig. 1A). Thus, invasive growth into the masseter and mylohyoid muscles was highly suspected. Additionally, a neighbouring submandibular lymph node was enlarged (Fig. 1B), and a small superficial biopsy was interpreted as ‘features consistent with desmoplastic fibroma’. Therefore, a hemimandibulectomy with excision of an enlarged lymph node was performed. The surgical specimen was solid and hard, with an ill-defined tumour in the mandible measuring 6.0×4.5×3.3 cm (Fig. 1C). Histologically, the lesion was composed of scirrhous fibrous tissue in which small strands and islands of tumour cells were often difficult to discern. There were single-file thin cords of inactive looking odontogenic epithelium, and these cells were cuboidal or polygonal shaped with bland nuclear features (Fig. 1D). The mitotic figures were inconspicuous throughout the tumour. In the focal areas, scattered clear cells were observed (Fig. 1E), and they were unreactive with the periodic acid–Schiff staining. Immunohistochemically, the tumour cells were highlighted by their diffuse and intense positivity for cytokeratin (CK) 5/6 (Fig. 1F), p40, p63, and CK19, but AE1/AE3 expression was variable. The average Ki-67 labelling index was approximately 15.9% in the highest part. Scattered epithelial clusters were observed in an extirpated lymph node; all were strongly positive for CK5/6 (Fig. 1G). Notably, several metastatic tumour nests formed the tubuloductal epithelial structures. Therefore, we diagnosed the patient with SOC with lymph node metastasis based on the overall findings. One year after the mandibulectomy, reconstructive surgery was performed. Later, the patient was closely followed, and local recurrence was identified 3 years later. A CT examination revealed cortical expansion and destruction of the mandibular symphysis. Furthermore, a recurrent tumour occupied the left infratemporal fossa (Fig. 2A,B), which was confirmed by coronal post-contrast fat-suppressed T1-weighted imaging (Fig. 2C). The recurrent tumour was resected at another institution, and chemo-radiotherapy was administered because of positive margins. Currently, the patient is in clinical remission. In the recurrent tumour, the histopathology essentially matched that of the primary SOC (Fig. 2D). However, some compressed tumour cells exhibited a unique reticular growth pattern (Fig. 2E), and the solid SOC nests were slightly larger. They lacked overt squamous and ameloblastic differentiation (Fig. 2F). As expected, we observed considerable perineural infiltration (Fig. 2G). Unlike the primary lesion, spindle cell proliferation, necrotic tumour nests, and myxoid stroma, although focal, were evident. Immunohistochemical evaluation revealed an identical phenotype to the primary and metastatic SOCs (Fig. 2H). In additional sections, the tumour expanded extensively to involve surrounding tissue (Fig. 3A), and there were multiple infiltrating nests of clear cells in a very dense collagenous stroma (Fig. 3B). To verify the diagnosis, fluorescence in situ hybridisation (FISH) analysis was performed to investigate a rearrangement of the EWSR1 gene. There was no evidence of either a EWSR1 split or EWSR1-ATF1 gene fusion in any of the primary and recurrent tumours, consistent with the diagnosis of SOC. Currently, SOC is defined as a low grade carcinoma with no metastatic potential.2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar, 3Soluk-Tekkesin M. Wright J.M. The World Health Organization Classification of odontogenic lesions: a summary of the changes of the 2022 5th ed.Turk J Pathol. 2022; 38: 168-184Google Scholar, 4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar However, it has become increasingly clear from the reported SOC cases that a malignant character is not always evident in the clinical setting.2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar The histological differential diagnosis of SOC is challenging in almost all cases.4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar Without cytological atypia, focal invasion into surrounding bone and muscle or intra- or peri-neural infiltration is the only criterion for diagnosing SOC as carcinoma.1Odell E.W. Koutlas I. Sclerosing odontogenic carcinoma.in: El-Naggar A.K. Chan J.K.C. Grandis J.R. WHO Classification of Head and Neck Tumours. 4th ed. IARC, Lyon2017: 209-210Google Scholar, 2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar, 3Soluk-Tekkesin M. Wright J.M. The World Health Organization Classification of odontogenic lesions: a summary of the changes of the 2022 5th ed.Turk J Pathol. 2022; 38: 168-184Google Scholar, 4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar, 5Ide F. Ito Y. Muramatsu T. et al.Sclerosing odontogenic carcinoma: a morphologic pattern or pathologic entity?.Oral Surg Oral Med Oral Pathol Oral Radiol. 2013; 115: 839Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar Given the presence of clear tumour cells in our case, we performed a FISH molecular genetic study for EWSR1 arrangement characteristic of clear cell odontogenic carcinoma.1Odell E.W. Koutlas I. Sclerosing odontogenic carcinoma.in: El-Naggar A.K. Chan J.K.C. Grandis J.R. WHO Classification of Head and Neck Tumours. 4th ed. IARC, Lyon2017: 209-210Google Scholar,2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar,4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar This was negative in the present lesion, as in other reported cases of SOC.2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar,4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar,6Todorovic E. Berthelet E. O'Connor R. et al.Sclerosing odontogenic carcinoma with local recurrence: case report and review of literature.Head Neck Pathol. 2019; 13: 371-377Crossref PubMed Scopus (19) Google Scholar, 7Seki-Soda M. Sano T. Ogawa I. et al.Two cases of odontogenic carcinoma with sclerosing features in the mandible: diagnostic difficulties in a sclerosing odontogenic carcinoma.J Oral Maxillofac Surg Med Pathol. 2020; 32: 149-155Crossref Scopus (1) Google Scholar, 8Seyiti P. Feng Y. Gao A. et al.An extensive sclerosing odontogenic carcinoma in mandible: a case report and literature review.Dentomaxillofac Radiol. 2020; 4920190426Crossref Google Scholar Besides the usual clear cell population, SOC can display unusual histomorphological patterns, including squamous differentiation, spindle cell foci, or ameloblastoma-like nests.6Todorovic E. Berthelet E. O'Connor R. et al.Sclerosing odontogenic carcinoma with local recurrence: case report and review of literature.Head Neck Pathol. 2019; 13: 371-377Crossref PubMed Scopus (19) Google Scholar,7Seki-Soda M. Sano T. Ogawa I. et al.Two cases of odontogenic carcinoma with sclerosing features in the mandible: diagnostic difficulties in a sclerosing odontogenic carcinoma.J Oral Maxillofac Surg Med Pathol. 2020; 32: 149-155Crossref Scopus (1) Google Scholar,9Hanisch M. Baumhoer D. Elges S. et al.Sclerosing odontogenic carcinoma: current diagnostic and management considerations concerning a most unusual neoplasm.Int J Oral Maxillofac Surg. 2017; 46: 1641-1649Abstract Full Text Full Text PDF Scopus (21) Google Scholar In the 2022 WHO classification, there was a brief comment that SOC may have infrequent islands of more epidermoid cells.4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar The lesion presented in this study showed a reticular arrangement of tumour cells, which has not been previously described.1Odell E.W. Koutlas I. Sclerosing odontogenic carcinoma.in: El-Naggar A.K. Chan J.K.C. Grandis J.R. WHO Classification of Head and Neck Tumours. 4th ed. IARC, Lyon2017: 209-210Google Scholar,2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar,4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar However, a nasopharyngeal squamous cell carcinoma case report described this unique growth pattern using the diagnostic terminology ‘reticular non-keratinising carcinoma’.10Petersson F. Vijayadwaja D. Loh K.S. et al.Reticular and myxoid non-keratinizing nasopharyngeal carcinoma: an unusual case mimicking a salivary gland carcinoma.Head Neck Pathol. 2014; 8: 364-368Crossref PubMed Scopus (10) Google Scholar The SOC tumour in our case also expressed CK5/6, p40, and p63; thus, SOC may be a form of primary intraosseous non-keratinising squamous cell carcinoma. Recently, Koutlas et al.11Koutlas I.G. Ponce K.J. Wazen R.M. et al.An unusual maxillary tumor with tubuloductal epithelial structures, solid epithelial nests and stromal odontogenic ameloblast-associated protein deposits. Tubuloductal/syringoid variant of central odontogenic fibroma with amyloid?.Head Neck Pathol. 2022; 16: 587-595Crossref Scopus (2) Google Scholar observed many duct-like epithelial structures in an amyloid variant of central odontogenic fibroma, suggesting ductal differentiation of neoplastic odontogenic epithelium. Evaluations of the lymph node metastasis in this study identified apparent tubular formation in the tumour nests, which has also been reported in at least three other cases.2Lim D. Tan C.C. Tilakaratne W.M. et al.Sclerosing odontogenic carcinoma - review of all published cases: is it a justifiable addition as a malignancy?.Braz J Otorhinolaryngol. 2022; 88: 118-129Crossref Scopus (3) Google Scholar The 2022 WHO classification clearly described that glandular differentiation may be seen in SOC.4Koutlas I.G. Sclerosing odontogenic carcinoma.in: WHO Classification of Tumours Editorial Board. Head and Neck Tumours. 5th ed. IARC, Lyon2022https://tumourclassification.iarc.who.int/chapters/52Google Scholar These observations support the odontogenic origin of SOC and also imply a histogenetic kinship between SOC and central odontogenic fibroma.12Tosios K.I. Giannoulis G. Kaklamanis L. et al.A central maxillary tumor with bland cytology, partly sclerotic stroma, and neural involvement. Scleroing odontogenic carcinoma or epithelial neurotropism in an odontogenic fibroma?.Oral Surg. 2022; 15: 624-629Crossref Scopus (2) Google Scholar In summary, we report the first case of SOC with recurrence and metastasis. The latter finding emphasises that SOC can be truly malignant. Therefore, awareness of the metastatic potential of SOC is essential in clinical practice. Furthermore, pathologists should be aware that the microscopic features of SOC significantly vary in the literature, from central odontogenic fibroma to clear cell odontogenic carcinoma, ameloblastic carcinoma or primary intraosseous carcinoma, not otherwise specified. We greatly thank Dr Rie Ohtomo and Dr Hirokazu Taniguchi of the Department of Clinical Laboratory, JR Tokyo General Hospital, for providing surgical specimens of a recurrent tumour; we would like to express our sincere gratitude to Dr Yukiko Sato and Dr Kengo Takeuchi of the Department of Pathology, Cancer Institute Hospital, for their significant contribution to molecular analyses. We also thank Editage (www.editage.com) for English language editing. This work was partially supported by research grants from the Sato Fund of the Nihon University School of Dentistry and by a grant from the Dental Research Center of the Nihon University School of Dentistry. The authors state that there are no conflicts of interest to disclosure." @default.
• W4361002039 created "2023-03-30" @default.
• W4361002039 creator A5031194703 @default.
• W4361002039 creator A5033171899 @default.
• W4361002039 creator A5036512670 @default.
• W4361002039 creator A5061923569 @default.
• W4361002039 creator A5062820568 @default.
• W4361002039 creator A5073195143 @default.
• W4361002039 date "2023-10-01" @default.
• W4361002039 modified "2023-10-13" @default.
• W4361002039 title "Sclerosing odontogenic carcinoma with local recurrence and lymph node metastasis" @default.
• W4361002039 cites W2163931943 @default.
• W4361002039 cites W2698815047 @default.
• W4361002039 cites W2897750599 @default.
• W4361002039 cites W2991197948 @default.
• W4361002039 cites W3016632343 @default.
• W4361002039 cites W3134327958 @default.
• W4361002039 cites W3192683316 @default.
• W4361002039 cites W4200571912 @default.
• W4361002039 cites W2053371697 @default.
• W4361002039 doi "https://doi.org/10.1016/j.pathol.2023.01.011" @default.
• W4361002039 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/37080825" @default.
• W4361002039 hasPublicationYear "2023" @default.
• W4361002039 type Work @default.
• W4361002039 citedByCount "1" @default.
• W4361002039 crossrefType "journal-article" @default.
• W4361002039 hasAuthorship W4361002039A5031194703 @default.
• W4361002039 hasAuthorship W4361002039A5033171899 @default.
• W4361002039 hasAuthorship W4361002039A5036512670 @default.
• W4361002039 hasAuthorship W4361002039A5061923569 @default.
• W4361002039 hasAuthorship W4361002039A5062820568 @default.
• W4361002039 hasAuthorship W4361002039A5073195143 @default.
• W4361002039 hasBestOaLocation W43610020391 @default.
• W4361002039 hasConcept C121608353 @default.
• W4361002039 hasConcept C126322002 @default.
• W4361002039 hasConcept C142724271 @default.
• W4361002039 hasConcept C2777546739 @default.
• W4361002039 hasConcept C2778125672 @default.
• W4361002039 hasConcept C2779013556 @default.
• W4361002039 hasConcept C2780849966 @default.
• W4361002039 hasConcept C2992571226 @default.
• W4361002039 hasConcept C71924100 @default.
• W4361002039 hasConceptScore W4361002039C121608353 @default.
• W4361002039 hasConceptScore W4361002039C126322002 @default.
• W4361002039 hasConceptScore W4361002039C142724271 @default.
• W4361002039 hasConceptScore W4361002039C2777546739 @default.
• W4361002039 hasConceptScore W4361002039C2778125672 @default.
• W4361002039 hasConceptScore W4361002039C2779013556 @default.
• W4361002039 hasConceptScore W4361002039C2780849966 @default.
• W4361002039 hasConceptScore W4361002039C2992571226 @default.
• W4361002039 hasConceptScore W4361002039C71924100 @default.
• W4361002039 hasIssue "6" @default.
• W4361002039 hasLocation W43610020391 @default.
• W4361002039 hasLocation W43610020392 @default.
• W4361002039 hasOpenAccess W4361002039 @default.
• W4361002039 hasPrimaryLocation W43610020391 @default.
• W4361002039 hasRelatedWork W2053411225 @default.
• W4361002039 hasRelatedWork W2341839242 @default.
• W4361002039 hasRelatedWork W2363109578 @default.
• W4361002039 hasRelatedWork W2369148130 @default.
• W4361002039 hasRelatedWork W2389855896 @default.
• W4361002039 hasRelatedWork W2472598939 @default.
• W4361002039 hasRelatedWork W2953716750 @default.
• W4361002039 hasRelatedWork W3031737811 @default.
• W4361002039 hasRelatedWork W4237682097 @default.
• W4361002039 hasRelatedWork W5509621 @default.
• W4361002039 hasVolume "55" @default.
• W4361002039 isParatext "false" @default.
• W4361002039 isRetracted "false" @default.
• W4361002039 workType "article" @default.