{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,5]],"date-time":"2026-01-05T14:46:53Z","timestamp":1767624413679,"version":"3.48.0"},"reference-count":24,"publisher":"MDPI AG","issue":"1","license":[{"start":{"date-parts":[[2026,1,4]],"date-time":"2026-01-04T00:00:00Z","timestamp":1767484800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Dentistry Journal"],"abstract":"<jats:p>Background: Primary intraosseous carcinoma (PIOC) is a rare and aggressive odontogenic malignancy that originates within the jaw bones without initial mucosal involvement. Its atypical and nonspecific symptoms frequently lead to diagnostic delays, especially in maxillary presentations. Methods: A 74-year-old male presented with persistent trigeminal-like neuralgic pain along the ophthalmic branch, initially misdiagnosed as secondary trigeminal neuralgia. MRI revealed a 45 \u00d7 46 \u00d7 34 mm mass occupying the right maxillary sinus with orbital wall destruction and dural invasion. Following histopathological confirmation of malignancy, a multidisciplinary team performed total maxillectomy with orbital exenteration and dural resection, followed by reconstruction using a temporoparietal flap. Adjuvant radiotherapy was administered. Results: Histopathology revealed invasive odontogenic carcinoma with atypical squamous features, dentinoid deposition, and perineural invasion. Postoperative recovery was uneventful, with complete pain resolution. MRI and PET surveillance over 2.5 years demonstrated no local recurrence. Conclusions: Maxillary PIOC may present exclusively with neuropathic pain, mimicking trigeminal neuralgia and leading to delayed diagnosis. In cases of unexplained facial pain with sinus or skull base involvement, odontogenic malignancies should be considered in the differential diagnosis. Early imaging and multidisciplinary management are key to achieving timely diagnosis, effective treatment, and improved quality of life.<\/jats:p>","DOI":"10.3390\/dj14010028","type":"journal-article","created":{"date-parts":[[2026,1,5]],"date-time":"2026-01-05T10:53:50Z","timestamp":1767610430000},"page":"28","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["When Cancer Mimics Pain: Maxillary Primary Intraosseous Carcinoma Misdiagnosed as Trigeminal Neuralgia"],"prefix":"10.3390","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-0481-2180","authenticated-orcid":false,"given":"Co\u0219arc\u0103","family":"Adina Simona","sequence":"first","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, George Emil Palade University of Medicine, Pharmacy, Science and Technology of Targu Mures, 540142 Targu Mures, Romania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sz\u00e1va","family":"Daniel","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, George Emil Palade University of Medicine, Pharmacy, Science and Technology of Targu Mures, 540142 Targu Mures, Romania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gherman Mircea","family":"Bogdan","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Emergency County Hospital T\u00e2rgu Mures, Gheorghe Marinesscu Street, nr 50, 540136 Targu Mures, Romania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mocanu","family":"Simona","sequence":"additional","affiliation":[{"name":"Department of Histology, Emergency County Hospital T\u00e2rgu Mures, Gheorghe Marinesscu Street, nr 50, 540136 Targu Mures, Romania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Petrovan","family":"Cecilia","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, George Emil Palade University of Medicine, Pharmacy, Science and Technology of Targu Mures, 540142 Targu Mures, Romania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8776-2654","authenticated-orcid":false,"given":"Mihai-Vlad","family":"Golu","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, George Emil Palade University of Medicine, Pharmacy, Science and Technology of Targu Mures, 540142 Targu Mures, Romania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ormeni\u015fan","family":"Alina","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, George Emil Palade University of Medicine, Pharmacy, Science and Technology of Targu Mures, 540142 Targu Mures, Romania"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2026,1,4]]},"reference":[{"key":"ref_1","unstructured":"WHO (2024). Head and Neck Tumours, International Agency for Research on Cancer, World Health Organization. [5th ed.]. WHO classification of tumours."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"406","DOI":"10.4103\/0973-029X.174615","article-title":"Primary Intraosseous Squamous Cell Carcinoma in Odontogenic Keratocyst: A Rare Entity","volume":"19","author":"Saxena","year":"2015","journal-title":"J. Oral Maxillofac. Pathol."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"608","DOI":"10.1007\/s12105-020-01234-z","article-title":"Primary Intraosseous Squamous Cell Carcinoma Involving the Jaw Bones: A Systematic Review and Update","volume":"15","author":"Carlan","year":"2021","journal-title":"Head Neck Pathol."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"25","DOI":"10.1186\/s12957-021-02465-2","article-title":"Primary Intraosseous Carcinoma in the Pediatric and Adolescent Mandible","volume":"20","author":"Oh","year":"2022","journal-title":"World J. Surg. Oncol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"320","DOI":"10.4103\/jomfp.JOMFP_25_16","article-title":"Primary Intraosseous Squamous Cell Carcinoma\u2014A Rare Odontogenic Malignancy","volume":"21","author":"Thakur","year":"2017","journal-title":"J. Oral Maxillofac. Pathol."},{"key":"ref_6","first-page":"794","article-title":"Primary Intraosseous Carcinoma of the Jaws","volume":"127","author":"Zwetyenga","year":"2001","journal-title":"Arch. Otolaryngol. Head Neck Surg."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"e71","DOI":"10.1016\/j.oooo.2012.05.019","article-title":"Solid-Type Primary Intraosseous Squamous Cell Carcinoma of the Mandible: A Case Report with Histopathological and Imaging Features","volume":"114","author":"Matsuzaki","year":"2012","journal-title":"Oral Surg. Oral Med. Oral Pathol. Oral Radiol."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"368504241305896","DOI":"10.1177\/00368504241305896","article-title":"Primary Intraosseous Squamous Cell Carcinoma Originating from a Mandibular Odontogenic Cyst: A Case Report","volume":"107","author":"He","year":"2024","journal-title":"Sci. Prog."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"189","DOI":"10.21147\/j.issn.1000-9604.2017.03.04","article-title":"Recurrence and Cancerization of Ameloblastoma: Multivariate Analysis of 87 Recurrent Craniofacial Ameloblastoma to Assess Risk Factors Associated with Early Recurrence and Secondary Ameloblastic Carcinoma","volume":"29","author":"Yang","year":"2017","journal-title":"Chin. J. Cancer Res."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"587","DOI":"10.5858\/arpa.2018-0417-SA","article-title":"Data Set for the Reporting of Malignant Odontogenic Tumors: Explanations and Recommendations of the Guidelines From the International Collaboration on Cancer Reporting","volume":"143","author":"Slootweg","year":"2019","journal-title":"Arch. Pathol. Lab. Med."},{"key":"ref_11","doi-asserted-by":"crossref","unstructured":"S\u00eerbu, I., Nisipasu, I.C., Savino, P., Custura, A.M., Radu, E.A., Nastasie, V., and S\u00eerbu, V.D. (2025). Surgical Management of a Maxillary Odontogenic Keratocyst: A Clinical Case Report. Dent. J., 13.","DOI":"10.3390\/dj13110514"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"131","DOI":"10.3892\/ol.2014.2644","article-title":"Primary Intraosseous Squamous Cell Carcinoma of the Maxilla Possibly Arising from an Infected Residual Cyst: A Case Report","volume":"9","author":"Sukegawa","year":"2015","journal-title":"Oncol. Lett."},{"key":"ref_13","first-page":"262","article-title":"Maxillary Carcinoma: A Wolf in Sheep\u2019s Clothing","volume":"55","author":"Mehanna","year":"2009","journal-title":"Can. Fam. Physician"},{"key":"ref_14","doi-asserted-by":"crossref","unstructured":"Bashir, A., Khan, Z.A., Maqsood, A., Prabhu, N., Saleem, M.M., Alzarea, B.K., Issrani, R., Khan, S.R., Ahmed, N., and Abbasi, M.S. (2023). The Evaluation of Clinical Signs and Symptoms of Malignant Tumors Involving the Maxillary Sinus: Recommendation of an Examination Sieve and Risk Alarm Score. Healthcare, 11.","DOI":"10.3390\/healthcare11020194"},{"key":"ref_15","doi-asserted-by":"crossref","unstructured":"Liu, Y., and Tanaka, E. (2025). Pathogenesis, Diagnosis, and Management of Trigeminal Neuralgia: A Narrative Review. J. Clin. Med., 14.","DOI":"10.3390\/jcm14020528"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"e120","DOI":"10.1055\/s-0039-3402034","article-title":"Skull Base Invasion Patterns of Malignant Head and Neck Tumors: A Neurosurgical Perspective","volume":"82","author":"Iwami","year":"2021","journal-title":"J. Neurol. Surg. B Skull Base"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"1806","DOI":"10.1111\/odi.13701","article-title":"Prevalence of Pain in Oral Cancer: A Retrospective Study","volume":"27","author":"Khawaja","year":"2021","journal-title":"Oral Dis."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"1033","DOI":"10.1080\/13803395.2019.1646711","article-title":"Effects of Acute Pain and Pain-Related Fear on Risky Decision-Making and Effort during Cognitive Tests","volume":"41","author":"Barnhart","year":"2019","journal-title":"J. Clin. Exp. Neuropsychol."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"878","DOI":"10.1007\/s10683-017-9515-6","article-title":"The Effect of Acute Pain on Risky and Intertemporal Choice","volume":"20","author":"Koppel","year":"2017","journal-title":"Exp. Econ."},{"key":"ref_20","doi-asserted-by":"crossref","unstructured":"Negrello, S., Pellacani, A., di Bartolomeo, M., Bernardelli, G., Nocini, R., Pinelli, M., Chiarini, L., and Anesi, A. (2020). Primary Intraosseous Squamous Cell Carcinoma of the Anterior Mandible Arising in an Odontogenic Cyst in 34-Year-Old Male. Reports, 3.","DOI":"10.3390\/reports3020012"},{"key":"ref_21","doi-asserted-by":"crossref","unstructured":"De Riu, G., Soma, D., Biglio, A., Raho, M.T., Mura, D., Michelon, F., Salzano, G., Piombino, P., Lechien, J.R., and Vaira, L.A. (2023). Primary Reconstruction of Total Maxillectomy with Custom-Made Subperiosteal Implant and Temporal Muscle Flap: A Case Report. Appl. Sci., 13.","DOI":"10.3390\/app13106269"},{"key":"ref_22","doi-asserted-by":"crossref","unstructured":"Fernandes, R. (2014). Local and Regional Flaps in Head and Neck Reconstruction: A Practical Approach, John Wiley & Sons. Available online: https:\/\/www.wiley.com\/en-ae\/Local+and+Regional+Flaps+in+Head+and+Neck+Reconstruction%3A+A+Practical+Approach-p-9781118947272.","DOI":"10.1002\/9781118947272"},{"key":"ref_23","first-page":"e69155","article-title":"Diagnosis and Prognostic Implications of Primary Intraosseous Carcinoma: A Case Report and Literature Review","volume":"16","author":"McDonald","year":"2024","journal-title":"Cureus"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"272","DOI":"10.1016\/S1470-2045(06)70620-9","article-title":"Treatment of Pulmonary Metastases from Primary Intraosseous Odontogenic Carcinoma","volume":"7","author":"Galetta","year":"2006","journal-title":"Lancet Oncol."}],"container-title":["Dentistry Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2304-6767\/14\/1\/28\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,5]],"date-time":"2026-01-05T11:18:11Z","timestamp":1767611891000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2304-6767\/14\/1\/28"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,4]]},"references-count":24,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,1]]}},"alternative-id":["dj14010028"],"URL":"https:\/\/doi.org\/10.3390\/dj14010028","relation":{},"ISSN":["2304-6767"],"issn-type":[{"value":"2304-6767","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,4]]}}}