Primary Intraosseous Carcinoma Research Articles

Invasion of the bucco-mandibular space by oral squamous cell carcinoma: histopathological analysis of invasion pattern.

This study aimed to determine the patterns of invasion of oral squamous cell carcinoma (OSCC) into the bucco-mandibular space (BMS) using detailed histopathological analysis and to assess clinical outcomes. Patients with OSCC who underwent segmental mandibulectomy or hemi-mandibulectomy combined with resection of the BMS between 2012 and 2021 were included. The invasions of the BMS were classified into three patterns. Pattern A was defined as a horizontal invasion, Pattern B as a vertical invasion, and Pattern C as an expansive invasion. In total, 109 patients were reviewed. Of these 109 patients, the primary tumor affected the lower gingiva in 78 patients, the buccal mucosa in 18 patients, and was a primary intraosseous carcinoma of the mandible in 13 patients. Invasion of the BMS was significantly associated with a higher pathological T stage, positive/close margins, and lower disease-free survival (DFS) rates. The DFS rates were 86.7% and 66.0% in the BMS non-invasion and invasion groups, respectively. The DFS rates for each type of invasion were 82.1% for Pattern A, 67.4% for Pattern B, and 48.0% for Pattern C (P=0.277). Patients with BMS invasion have a poorer prognosis than those without invasion of the BMS. Therefore, adjuvant therapy is necessary, especially in Patterns B and C. Evaluation of preoperative BMS invasion patterns is important for predicting the prognosis of OSCC.

Primary intraosseous carcinoma arising in a dentigerous cyst: A case report

AbstractDentigerous cysts are among the most common cysts of the jaw. They are attached to the crown of an unerupted tooth, most frequently third molars and maxillary canines. They are often asymptomatic and may therefore be diagnosed late. We report a case of a primary intraosseous carcinoma arising from the wall of a dentigerous cyst and describe the expression of typical genes associated with tumour growth and prognosis. The tumour expressed cytokeratins (CK) 5/6, 8, 14 and 19, but not CK7. Ki‐67 and P53 were upregulated in dysplastic epithelium and tumour tissue. Although rare, the possibility for tumour development in the wall of a common cyst should be considered. The case emphasizes the importance of histopathological examination of surgical specimens, even if the clinical diagnosis seems obvious.

PRIMARY INTRAOSEOUS CARCINOMA - CASE REPORT

A 65-year-old patient, melanoderma, smoker since 10 years old, came to the oral and maxillofacial service complaining of pain in the jaw on the right side for one year. The intraoral physical examination revealed an ulcerated, leukoplasic and erythematous lesion in the region of the right posterior alveolar ridge. Imaging exams showed a radiolucent, unilocular lesion, with a well-circumscribed, radiopaque halo in the posterior mandibular region on the right side. Hematological examination was requested and an incisional biopsy was performed. The histopathology revealed a fragment of malignant neoplasm of epithelial origin characterized by the proliferation of tumor cells arranged in islands growing inside the connective tissue and a fragment of a cystic lesion of odontogenic origin characterized by the presence of a pathological cavity lined by stratified epithelium, with a consistent diagnosis of primary intraosseous carcinoma. The patient was referred to the oncology service. A 65-year-old patient, melanoderma, smoker since 10 years old, came to the oral and maxillofacial service complaining of pain in the jaw on the right side for one year. The intraoral physical examination revealed an ulcerated, leukoplasic and erythematous lesion in the region of the right posterior alveolar ridge. Imaging exams showed a radiolucent, unilocular lesion, with a well-circumscribed, radiopaque halo in the posterior mandibular region on the right side. Hematological examination was requested and an incisional biopsy was performed. The histopathology revealed a fragment of malignant neoplasm of epithelial origin characterized by the proliferation of tumor cells arranged in islands growing inside the connective tissue and a fragment of a cystic lesion of odontogenic origin characterized by the presence of a pathological cavity lined by stratified epithelium, with a consistent diagnosis of primary intraosseous carcinoma. The patient was referred to the oncology service.

A rare case of primary intraosseous carcinoma of the mandible with perineural extension mimicking neurolymphomatosis on [18F]-FDG PET/CT.

A rare case of primary intraosseous carcinoma of the mandible with perineural extension mimicking neurolymphomatosis on [18F]-FDG PET/CT.

Comparison of maximum and mean standardized uptake values of jaw pathologies with bone SPECT/CT: an especial focus on medication-related osteonecrosis of the jaw.

To investigate the comparison of maximum and mean standardized uptake values (SUVs) of jaw pathologies with bone Single-photon emission computed tomography/computed tomography (SPECT/CT), and a special focus on medication-related osteonecrosis of the jaw (MRONJ). Eighty-nine patients with jaw pathologies (63 MRONJ, 13 chronic osteomyelitis, 11 osteoradionecrosis and 2 primary intraosseous carcinoma) underwent bone SPECT/CT scans acquisition at 4 h after intravenous injection of Tc-99m hydroxymethylene diphosphonate in this prospective study. The evaluation of mean and maximum SUVs of jaw pathologies were performed using Q. Metrix and Xeleris workstation and defined the data automatically. Statistical analyses were performed by Pearson's correlation coefficient for comparison of maximum and mean SUVs and Mann-Whitney U-test for SUVs of MRONJ. A P value lower than 0.05 was considered to indicate statistical significance. Maximum SUVs of MRONJ, chronic osteomyelitis, osteoradionecrosis and primary intraosseous carcinoma were 17.6 ± 8.4, 21.7 ± 7.1, 11.9 ± 4.8 and 26.6 ± 7.0, respectively. Mean SUVs of MRONJ, chronic osteomyelitis, osteoradionecrosis and primary intraosseous carcinoma were 10.1 ± 4.9, 11.9 ± 3.3, 7.0 ± 2.8 and 10.1 ± 4.5, respectively. The maximum SUV of jaw pathologies was significantly correlated with the mean SUV (Y = 0.494X + 1.228; R2 = 0.786; P < 0.001). Furthermore, maximum and mean SUVs of MRONJ had significant differences in underlying diseases, medication and staging. The maximum and mean SUVs with bone SPECT/CT can be an effective tool for the quantitative evaluation of jaw pathologies, especially MRONJ.

Malignant odontogenic tumors. A report of a series of 30 cases and review of the literature

Malignant odontogenic tumors are rare entities and can arise de novo or from malignant transformation of pre-existing lesions. The aim of the present study was to report 30 cases of malignant odontogenic tumors diagnosed at a single diagnostic center over a 38-year period. Materials and Methods: All cases of malignant odontogenic tumors diagnosed between 1980 and 2018 were retrieved from the files of the School of Dentistry, University of Buenos Aires; their frequency and clinical, radiographic, and histopathological features were analyzed. The obtained data were compared with the information reported in the literature. Results: Malignant odontogenic tumors accounted for 2 % of odontogenic tumors and 0.05 % of all oral-maxillofacial diseases. Mean age was 43 years (8 to 87 years). No differences in sex distribution were observed. The most frequent location was the mandible, mostly the posterior mandibular body and the ramus. Odontogenic carcinomas were the most frequent tumors, and primary intraosseous carcinomas were the most common in this group. Conclusions: This is the largest case series of malignant odontogenic tumors diagnosed at a single center reported in the literature to date. Diagnosis of these tumors is challenging based on their rarity and complexity and relies on careful analysis of the histopathological, clinical, and radiographic features.

Primary Intraosseous Carcinoma, Not Otherwise Specified with Ameloblastic Carcinoma Features in a Middle-aged Woman

Primary Intraosseous Carcinoma, Not Otherwise Specified (PIOC, NOS) is an extremely rare neoplasm defined as squamous cell carcinoma in the mandible, presumably from the residue of odontogenic epithelium. The diagnosis is often challenging, and the prognosis is poor. The treatment of choice is surgical excision but usually declined for cosmetic reasons. A 41-year-old Melanesian woman with a mass in her right jaw since two years ago but rapidly growing in the last six months, accompanied with pain. Irregular hard mass was found in the right mandible. Radiology studies revealed lytic tumors, likely infiltrating. No metastasis was found. The first biopsy revealed acanthomatous ameloblastoma, and the second biopsy revealed pseudoepitheliomatous hyperplasia. Wide excision was then conducted with pathology results of PIOC and NOS with some features of ameloblastic carcinoma. This case may arise from a previous benign precursor. Further genetic information may be needed. The treatment of choice is surgical excision. The prognosis needs further follow-up. Primary intraosseous carcinoma, not otherwise specified (PIOC, NOS) merupakan keganasan odontogenik yang langka. Diagnosisnya sulit dengan prognosis buruk. Tata laksana utama adalah bedah eksisi yang cenderung ditolak karena alasan estetik. Seorang perempuan ras Melanesia usia 41 tahun, mengeluh benjolan rahang kanan sejak dua tahun dan makin membesar terutama 6 bulan terakhir disertai nyeri. Massa tidak teratur dengan batas tidak jelas di mandibula kanan dengan ukuran 10 x 8 x 4 cm. Pemeriksaan radiologis menegaskan massa infiltratif, tetapi tidak menemukan anak sebar. Biopsi awal menunjukkan ameloblastoma, biopsi selanjutnya menunjukkan pseudoepitheliomatous hyperplasia. Eksisi luas dilakukan dan patologi menemukan PIOC, NOS dengan sebagian ciri ameloblastic carcinoma. Patologi dari tiga biopsi menunjukkan massa jinak odontogenik yang mengalami inflamasi kronis kemudian menjadi ganas. Prognosis masih belum bisa dipastikan.

AGGRESSIVE PRIMARY INTRAOSSEOUS CARCINOMA AMELOBLASTOMA-LIKE INVOLVING ANTERIOR REGION AND MANDIBULAR BODY: CASE REPORT

Primary intraosseous carcinoma of the jaw is a rare malignant tumor with poor prognosis. A 66-year-old male smoker presented with swelling in the anterior region and right body mandibular, with extraoral fistula and palpable cervical lymph nodes. Intraoral examination evidenced an ulceration with a hard consistency. Panoramic radiograph and computed tomography revealed a large lesion with bone destruction in the region. After incisional biopsy, microscopic examination revealed a proliferation of epithelial cells sometimes similar to squamous cells, forming keratin pearls, or similar to the odontogenic epithelium, arranged in nests and islands of basaloid cells with peripheral palisading. Atypia, pleomorphism, and mitoses were observed, leading to the diagnosis of primary intraosseous carcinoma ameloblastoma-like. The patient underwent mandibulectomy, partial glossectomy, and removal of the floor of the mouth muscles. Follow-up is still being carried out and 1 month after surgery the patient is in stable condition without pain.

Outcome of primary intraosseous carcinoma: Case review of a single institution.

The purpose of this study was to investigate the prognostic factors and treatment of primary intraosseous carcinoma (PIOC). Patients diagnosed with POIC and received treatment in Sichuan Cancer Hospital from 2000 to 2019 were followed up and retrospectively reviewed. A total of 28 patients were included in the study, with a mean age of 60years (60±10.11). The 2-year and 5-year overall survival (OS) were 60.7% and 38.5%, respectively. In the univariate analysis, surgery combined with adjuvant therapy improved the OS compared with surgery or radiotherapy alone (p=0.035), and patients who received postoperative adjuvant radiotherapy had a higher OS than those who received radical radiotherapy (p=0.01). In addition, patients with well-differentiated tumors have increased progression-free survival (p=0.01). Multivariate analyses showed that radiotherapy was an independent indicator for OS (p=0.007). Surgery combined with adjuvant therapy is the superior treatment strategy for PIOC at present. This study is the first to confirm the positive role of radiotherapy in treating PIOC with data to back it up.

Primary intraosseous squamous cell carcinoma arising in odontogenic cysts: A report of five cases and a review of the literature

Primary intraosseous carcinoma (PIOC) is rare entity with an estimated incidence of 0.01-0.02% of all oral cancer. To date, 133 PIOC cases have been reported with most occurring in the setting of periapical/residual cysts. Distinc- tion from carcinomas arising from the oral or antral mucosa may be difficult, especially when the cortical plates have been destroyed. Therefore, signs of oral ulceration preclude the diagnosis of PIOC. Identification of a benign cystic structure exhibiting transition to a squamous cell carcinoma may be of help in establishing the diagnosis and etiology of the tumor. The aim of our study was to evaluate the cases of only primary intraosseous squamous cell carcinomas arising in odontogenic cysts diagnosed in the Oral Pathology Laboratory at New York Presbyterian Queens between 1995-2019 with the goal of adding valuable information to the literature for this uncommon en- tity. Five cases of PIOC were identified. For each case the histology, demographics, radiographic descriptions and radiographs when possible were reviewed. In all cases, transition from a cystic structure to infiltrating carcinoma was identified. In no case did the overlying surface mucosa exhibit dysplastic changes or transition to carcinoma. Four cases occurred in the maxilla and one in the mandible. The average age of patients in this study was 67.6 years with a range of 37 to 81. Three patients were males and two were female. Three cases presented as ill-defined radiolucencies and one was described as a cystic expansion. Two PIOCs arose in the setting of a periapical cyst, one in the setting of a residual cyst and one in the setting of a dentigerous cyst. Two cases had associated bone loss. While uncommon, the possibility of carcinoma arising in what appears clinically to be a cyst, supports the need to submit all tissue for microscopic examination.

Primary intraosseous squamous cell carcinoma arising in odontogenic cysts: a series of three cases with immunohistochemical analysis

<h3>Introduction</h3> Carcinomatous transformation in odontogenic cysts is rare, with few cases described in the literature. Squamous cell carcinoma (SCC) is the most common form of malignant transformation in odontogenic cysts. Radicular and dentigerous cysts are the most common odontogenic cysts to undergo malignant transformation. We present three cases of primary intraosseous carcinoma (PIOC) arising in odontogenic cysts. <h3>Method and Materials</h3> Following IRB approval, cases were identified from the University of Florida Biopsy Service archives (1994-2020). The clinical and demographic data was reviewed, along with radiographs and original slides. An immunohistochemistry (IHC) panel including CK19, p53, and Ki-67 was performed on all cases. <h3>Results</h3> The first case occurred in a 67-year-old male with a radiolucency around impacted #17. Microscopically, a verrucous carcinoma was diagnosed arising within a developmental odontogenic cyst, most likely an orthokeratinized odontogenic cyst. The second case involved a 59-year-old male with a lesion adjacent to tooth #15. Histologic examination revealed a well differentiated SCC arising from an odontogenic keratocyst (OKC). The final case involved a 13-year-old male with a radiolucency near teeth #6-7. A well differentiated SCC was diagnosed arising from an OKC. Further investigation to assess for Nevoid Basal Cell Carcinoma Syndrome was recommended for this case, but the outcome was unavailable. IHC analysis revealed P53 positivity for all cases. Ki67 showed 80-90% positivity in the basal layer of the odontogenic epithelium for cases 1 and 3, and 50% positivity for case two. Ck19 was only focal positivity in all cases. <h3>Conclusion</h3> We present three cases of squamous cell carcinoma arising within odontogenic cysts. P53 and Ki-67 positivity aid in confirmation of the aggressive nature of each tumor. CK19 staining was weak, despite the obvious odontogenic nature of the epithelium in all cases. Careful follow-up for odontogenic cysts demonstrating recurrence or progressive growth is imperative.

FNAC Diagnosis of Primary Intraosseous Squamous Cell Carcinoma Mandible: A Rare Case Report

Primary intraosseous squamous cell carcinoma of jaw is a rare tumour. It arises from the lining of an odontogenic cyst or de novo from postulated odontogenic cell rests and is rarely diagnosed by FNAC. The primary intraosseous carcinoma of squamous cell origin is very infrequent and only few cases are reported in available literature. The following case report describes a case of primary intraosseous squamous cell carcinoma of mandible in a 45 year old male patient with emphasis on cytological features. The main purpose is to highlight diagnostic significance of FNAC for rapid and accurate diagnosis of lesions of mandible and unusual clinical presentation.

Primary intraosseous carcinoma in the pediatric and adolescent mandible

BackgroundPrimary intraosseous carcinoma (PIOC) is a rare malignant odontogenic tumor that predominantly occurs in males older than 50 years. PIOC can be misdiagnosed as odontogenic cyst because it occasionally shows a well-defined border on radiography. In this study, related literatures of pediatric and adolescent PIOC cases were analyzed under strict PRISMA guidelines along with an adolescent case who was provisionally misdiagnosed as an odontogenic cyst.MethodsAll case reports for PIOC published in English from 1966 to 2021 were collected. Cases under the age of 20 were classified as pediatric and adolescent populations in this study. A total of 12 pediatric and adolescent cases including 11 PIOCs from the literature and one new case of a 14-year-old female were analyzed. Clinical and radiographic features, diagnosis and treatment approaches, and prognosis were investigated.ResultsAges ranged from 4 to 18 years. The female to male ratio was 1.4:1. Seven cases occurred in the mandible. Swelling was observed in 11 patients. The radiologic borders were well-defined in six cases and corticated in four cases. Tooth displacement and root resorption were observed in four and six cases, respectively. The provisional diagnosis for seven patients was odontogenic cyst and enucleation was performed in six cases including the new case. During the follow-up period, local recurrence occurred in three patients. The pediatric and adolescent PIOC cases with local recurrence showed poor prognosis. The locally recurred lesion in the new case did not decrease in size despite concurrent chemo-radiation therapy.ConclusionsThree-dimensional imaging modalities and incisional biopsy with multiple specimens are necessary to rule out PIOC in the lesions with atypical radiographic findings. PIOC should be diagnosed differentially from odontogenic cyst even in pediatric and adolescent populations to properly manage the disease with poor prognosis.

A case of primary intraosseous carcinoma of the mandible with functional restoration by dental implants after immediate reconstruction using sagittal split sliding osteotomy and coronoid process grafting

A case of primary intraosseous carcinoma of the mandible with functional restoration by dental implants after immediate reconstruction using sagittal split sliding osteotomy and coronoid process grafting

53 A Rare Malignant Lesion of the Jaws

Abstract Introduction Primary intraosseous carcinoma (PIOC) of the jaw is an incredibly rare lesion with approximately 200 cases reported in the literature. PIOC are mostly asymptomatic incidental radiographic findings and present as poorly-defined, non-corticated radiolucencies often with root resorption and cortical perforation. Prognosis is considered poor and best predicted by histological grade. Case report A gentleman in his 50s presented to his dentist with a draining sinus from his LR6 tooth. A radiograph revealed a non-corticated radiolucency localised to the distal root of the LR6. 7 months later, he returned with pain and mobility of the LR7. Another radiograph revealed a non-corticated radiolucency from around the distal root of the LR6 extending to the LR8. Urgent biopsy of the mandibular bone was arranged by the oral and maxillofacial surgery team. Histopathology revealed the lesion was likely a benign squamous odontogenic tumour. However, it was not possible to exclude a well-differentiated squamous cell carcinoma. Correlation of clinical findings, imaging and multidisciplinary team discussion was recommended. Following the MDT meeting, excision of the bony lesion was undertaken. Histopathology this time returned as primary intraosseous carcinoma NOS T4N0M0, thought to have arisen from a radicular cyst associated with the LR6/7. The treatment was right segmental mandibulectomy, fibular free flap, selective neck dissection (I-III) and adjuvant radiotherapy. Conclusions This case highlights how subtle radiographic features can represent malignancy in lesions appearing cystic at first. Although rare, clinicians should be aware of PIOC as a differential diagnosis when presented with radiographic features which could represent malignancy.

English

Primary intraosseous carcinoma (PIOC) previously named as primary intraosseous squamous cell carcinoma (PIOSCC), derived from an odontogenic keratocyst (OKC) is a locally aggressive rare malignant neoplasm of the jaws with poor prognosis. The incidence of carcinomas arising from odontogenic cysts was reported to be approximately 1 – 2 / 1000. The knowledge of the histopathological features of PIOC allows accurate and early diagnosis of the lesion so that an early and appropriate treatment can be instituted for better prognosis. Primary intraosseous squamous cell carcinoma (PIOSCC) is a carcinoma arising from the central bone without any initial connection to various epithelia.1 In 2017, according to WHO classification of tumours, it was renamed as primary intraosseous carcinoma (PIOC).2 According to the current WHO classification, PIOC is a central jaw carcinoma that cannot be classified as any other condition and presumably has developed from odontogenic cysts or other benign precursors. The most common odontogenic cysts having a significant probability of malignant transformation are dentigerous cysts, residual periapical cysts, and odontogenic keratocysts (OKCs).3,4 The transition from this benign cystic lining to squamous cell carcinoma may remain clinically undetected, and the delay in diagnosis has a significant influence on treatment and prognosis.5 Therefore, the initial radiographic features of PIOC arising from an OKC (PIOC ex - OKC) are similar to those of benign odontogenic tumours or cysts, including conventional OKCs.4 Hence, it is mandatory to further investigate it histopathologically for final diagnosis and proper treatment plan. Malignant changes in OKCs have been rarely reported. To the best of our knowledge, very few cases of PIOC ex - OKC have been described in the literature. The purpose of this article is to report a case of PIOC that originated from an OKC and to shed light on the process of malignant transformation of this rare lesion. Here, is the case report describing an extremely rare case of PIOC of the maxilla derived from an OKC in a 45 - year - old female patient.

Response to ‘Primary intraosseous carcinoma, NOS arising from a dentigerous cyst: A case report’

Response to ‘Primary intraosseous carcinoma, NOS arising from a dentigerous cyst: A case report’

Primary intraosseous carcinoma, NOS arising from a dentigerous cyst: A case report

AbstractIntroductionPrimary intraosseous carcinoma, NOS (PIOC), is a rare malignancy which has been described in the maxilla and mandible often arising from odontogenic cysts. The authors describe a case of PIOC arising from a dentigerous cyst associated with a lower right third molar in 78‐year‐old male.AimsTo demonstrate that seemingly innocuous pathology may represent a malignant process. In the presence of ‘red‐flag’ symptoms, one must consider alternative diagnoses and action them accordingly.Findings and DiscussionThe initial clinical and radiographic features suggested a common innocuous presentation, although other features which presented later were suggestive of malignancy. PIOC is treated conventionally by radical surgery to achieve a disease‐free margin with adjuvant neck dissection. In the case described, the patient underwent hemi‐mandibulectomy with neck dissection and fibula free flap reconstruction.ConclusionPathology associated with third molars is extremely common, very occasionally malignancy can mimic these more common conditions. As such, careful clinical examination of the affected site and neighbouring structures (including the lymph nodes of the head and neck) should be routinely performed in all patients. Even in the presence of typical features clinical and radiographic features, tissue samples should be sent for histopathological analysis of what may appear to be harmless follicular or early cystic tissue.

Primary Intraosseous Carcinoma of the Mandible: A Case Report

Primary intraosseous carcinoma (PIOC) is a rare tumor that has been infrequently reported. Carcinoma arising primarily from the jaw is a locally aggressive lesion with poor prognosis. Primary intraosseous carcinoma (PIOC) lesion develops either de novo remnants of odontogenic epithelium, odontogenic cyst/tumor, epithelium remnants, or/and salivary gland residues. This article describe a case of primary intraosseous carcinoma of mandible.

Risk factors for distant metastasis in locoregionally controlled oral squamous cell carcinoma: a retrospective study

The control of distant metastasis in oral squamous cell carcinoma is an important determinant of improved prognosis. The study aimed to identify risk factors for distant metastasis in patients with locoregionally controlled oral carcinoma. We identified 982 patients with oral squamous cell carcinoma treated at our hospital between January 2008 and December 2017. After excluding patients with distant metastasis at initial treatment, patients with metastasis to the oral cavity, those receiving palliative treatment, and those lacking follow-up data, 941 patients were selected. Finally, among these 941 patients, 887 with locoregionally controlled oral squamous cell carcinoma were included in the study. Among the 887 patients, 36 had confirmed distant metastasis (4.1%), and the lung was the most common site (31/36 patients, 86.1%). Multivariate analysis showed that the incidence of primary intraosseous carcinoma of the mandible, cervical lymph node metastasis at levels IV and V, and the presence of pathological extranodal extension were significant risk factors for distant metastasis. When treating patients with oral squamous cell carcinoma who are positive for the aforementioned risk factors, the possibility of developing distant metastases must be accounted for, and aggressive treatment should be planned accordingly.