Primary Intraosseous Squamous Cell Carcinoma Research Articles

Primary intraosseous squamous cell carcinoma presenting as lytic lesion in mandible: Common presentation of an uncommon illness

Primary intraosseous squamous cell carcinoma presenting as lytic lesion in mandible: Common presentation of an uncommon illness

Differential Retrospective Analysis in Oral Cancerous, Pre-cancerous, and Benign Tissue Biopsies.

Background/AimOral epithelia demonstrate a broad spectrum of pre-cancerous, cancerous, and benign lesions. The aim of this study was to record and analyze the prevalence of various oral and intraosseous lesions, highlighting malignancies that are hard to clinically identify as such too.Materials and methodsA series of 536 oral lesions were collected covering a period of 8.5 years. Epidemiological and clinico-histopathological data were stratified and analyzed retrospectively.ResultsAccording to extensive differential analysis, the male to female ratio for oral squamous cell carcinoma was estimated at 1:1, for pre-cancerous lesions at 1:2, and for lichen planus at 1:5. The prevalent diagnostic category were cysts (n = 223, 41.6%). The biological behavior of lesions differed among anatomic sites (P<0.001). Concordance between clinical suspicion of pre-cancerous or malignant lesions and histological verification was 96.4% (P<0.001).ConclusionsPrimary intraosseous squamous cell carcinoma, acinic cell carcinoma, clear cell myoepithelial carcinoma, aggressive osteoblastoma/parosteal osteosarcoma, and undifferentiated carcinoma raised no clinical suspicion of malignancy reflecting the importance of training in oral biopsy taking.

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.

Need for Panel of Immunohistochemical Markers in Primary Intraosseous Squamous Cell Carcinoma Ex Odontogenic Keratocyst

Need for Panel of Immunohistochemical Markers in Primary Intraosseous Squamous Cell Carcinoma Ex Odontogenic Keratocyst

A primary intraosseous squamous cell carcinoma case report and literature review. The new WHO classification

AbstractAimCyst‐like lesions in the mandible rarely develop into malignancies, and the reported incidence is between 0.3% and 2%. The present study describes a rare case of primary intraosseous squamous cell carcinoma of the mandible arising from an odontogenic cyst.Materials and MethodsAn 80‐year‐old male was referred to Trieste University Maggiore Hospital (Trieste, Italy), with acute pain in the left retromolar area.An initial examination revealed extra oral swelling without paresthesia of the IAN. Following an intraoral examination, the oral mucosa was edematous, percussion pain was experienced on the lower left second molar. Panoramic radiography revealed a retained lower left wisdom tooth and an irregular radiolucent area between the lower left second molar and the mandibular angle with clear margins. Computed tomography revealed diffuse bone resorption and an extensive loss of cortical bone on the lingual side.ResultsA biopsy was performed during the surgery to remove the second lower left molar and the wisdom tooth, the pathological diagnosis was of squamous cell carcinoma arising from the epithelial lining of the odontogenic cyst. MRI with contrast agent was also performed. Shortly after the patient has been proposed a complete resective surgery of the mass including the infiltrated areas and then a reconstructive part to restore the function and aesthetic.ConclusionThe follow‐up of a large inflammatory cyst is essential for early diagnosis of malignant neoplasm. This approach allows lower invasive treatments and major survival of these patients.

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.

Intraosseous Squamous Cell Carcinoma Associated with Denosumab-Induced Osteonecrosis of the Jaw

Medication-related osteonecrosis of the jaw (MRONJ) has been associated with the use of different drugs administered in the treatment of malignant neoplasms or metastases and in antiresorptive therapies. Since 2010, denosumab, a monoclonal antibody whose mechanism of action is to prevent the activation of the RANK receptor on the surface of osteoclasts, has been associated with osteonecrosis of the jaw. Primary intraosseous squamous cell carcinoma (PIOSCC) is a rare neoplasm characterised by the presence of squamous cells within the maxillary or mandibular bone without an initial clinical association with the oral mucosa. We present a case of a PIOSCC in an osteonecrosis of the jaw related to a patient who received antiresorptive treatment with denosumab outlining the clinical and histopathological features of MRONJ and PIOSCC.

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.

Sequential treatment from mandibulectomy to reconstruction on mandibular oral cancer - Case review I: mandibular ramus and angle lesion of primary intraosseous squamous cell carcinoma.

Primary intraosseous squamous cell carcinoma (PIOSCC) is very rare type of squamous cell carcinoma (SCC) that occurs within the jaw and arises from remnants of odontogenic epithelium with no connection to the oral mucosa. This study reports two cases of PIOSCC of the mandible. Reported in this article are two cases of PIOSCC of the mandible that were treated with resection and reconstruction using a fibular free flap. The first case was a 36-year-old male patient who complained of right mandibular pain. Computed tomography (CT) and panoramic radiograph revealed a large radiolucency in the mandibular ramus area. At first, an odontogenic keratocyst was tentatively diagnosed, and an excision procedure was carried out at another clinic. A final biopsy after cyst enucleation revealed well-differentiated SCC, so we proceeded with segmental mandibulectomy and reconstruction using a fibular free flap. The second case was a 48-year-old male patient with left mandibular pain. CT and panoramic radiograph revealed irregular radiolucency in the mandibular angle area near tooth #38. At first, osteomyelitis was tentatively diagnosed, and a curettage was carried out. A later biopsy revealed well-differentiated SCC, so segmental mandibulectomy and reconstruction with a fibular free flap were secondarily performed. Our two cases have had no recurrence. The facial appearance of both patients is satisfactory, and the neo-mandibular body created using a fibular bone transfer displays adequate bony volume.

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

ObjectivesResidual 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 methodsSixty-four histopathologically confirmed residual cysts were analysed based on their clinical, radiological and histopathological features. Their management and follow-up were also noted.ResultsThe 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.ConclusionResidual 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.

Primary Intraosseous Squamous Cell Carcinoma Involving the Jaw Bones: A Systematic Review and Update.

Primary intraosseous oral squamous cell carcinoma (PIOSCC) is a rare malignant neoplasm that affects the jaws. Despite its aggressive biological behavior, there are no studies that evaluated the clinicopathological features of this tumor and parameters associated with its prognosis. The objective of the present study was to conduct a systematic review of the available data on oral and maxillofacial PIOSCC in order to determine its clinicopathological characteristics and biological behavior. We conducted a systematic review in May 2020 in multiple databases using a specific search strategy. Cases diagnosed as PIOSCC in the oral cavity and maxillofacial complex that had sufficient histopathological data, absence of ulceration in the oral mucosa, a negative result for a distant primary tumor, and radiographic evidence of an osteolytic lesion that was entirely or mostly surrounded by the jaw bones were included. A total of 109 published articles were included in our systematic review, corresponding to 257 cases. PIOSCC showed a male predilection (69.3%) and a preference for the mandible (7:1), with the posterior region being the most commonly affected site. The mean age at diagnosis was 57.3years. Cortical expansion, pain, and lip/facial paresthesia were the most common clinical features. Regarding histopathological features, most PIOSCC were well-differentiated and the solid subtype was the most common. Statistical analysis showed that PIOSCC located in the mandible (p = 0.03) and recurrence (p < 0.01) were significantly associated with a higher mortality rate. PIOSCC has a poor prognosis, with high rates of mortality.

Primary intraosseous squamous cell carcinoma arising from an odontogenic keratocyst: case series and literature review.

Background The aim of this study was to investigate the clinicopathologic features of primary intraosseous squamous cell carcinoma arising from an odontogenic keratocyst (PIOSCC ex OKC) and comprehensively improve the understanding of this disease. Material and Methods We retrospectively investigated five cases of PIOSCC ex OKC at Peking University School and Hospital of Stomatology. We also conducted a systematic review of studies on PIOSCC ex OKC by using online databases from their inception until February 2020. Results In our series of five cases, all lesions were located in the mandible. Three cases (60%) showed recurrent OKCs and two cases (40%) showed primary OKCs. During the follow-up period, one patient died of local relapse. No patients developed metastasis. On the basis of our literature survey, we selected 22 articles reporting 29 patients with PIOSCC ex OKC. Seven of these patients (24.1%) showed local recurrence, three patients (10.3%) developed cervical metastasis, three patients (10.3%) developed distant metastasis (in the pleura in one case and in the lung in two cases), and seven patients died from the disease during the follow-up period. The disease-specific 5-year survival rate in the study group was 53.2%. Through univariate and multivariate analysis, local recurrence was identified as the only significant independent prognostic factor for survival (P < 0.05). Conclusions The results suggest that PIOSCC ex OKC is a rare intermediate-grade malignancy. Although elective neck dissection is typically unnecessary, adequate therapy should be applied to achieve the lowest local recurrence rate possible to ensure a favorable survival rate. Key words:Primary intraosseous squamous cell carcinoma, odontogenic keratocyst, prognosis.

PRIMARY INTRAOSSEOUS SQUAMOUS CELL CARCINOMA OF THE MANDIBLE: A RARE CASE REPORT

The primary intraosseous squamous cell carcinoma (PISCC) is a rare malignant neoplasm of epithelial origin that affects the jaw bone interior without affecting the oral mucosa in a primary way. A 66-year-old male patient presented an intraoral volume increase, with coloring similar to the mucosa, hardened consistency, and painful symptoms located in the mandibular body left side. The radiographic examination showed a unilocular radiolucent image with poorly defined limits associated with the mandibular fracture. An incisional biopsy was performed, and the histopathologic examination demonstrated a malignant neoplasm of epithelial origin characterized by the proliferation of pleomorphic cells organized in sheets, nests, and cords and were also dispersed in a fibrous connective tissue stroma. Malignant neoplasms were discarded in other sites, and the diagnosis of PISCC was established. The patient underwent complete surgical resection of the lesion and is currently being followed up after 6 months without clinical-radiographic signs of relapse. The primary intraosseous squamous cell carcinoma (PISCC) is a rare malignant neoplasm of epithelial origin that affects the jaw bone interior without affecting the oral mucosa in a primary way. A 66-year-old male patient presented an intraoral volume increase, with coloring similar to the mucosa, hardened consistency, and painful symptoms located in the mandibular body left side. The radiographic examination showed a unilocular radiolucent image with poorly defined limits associated with the mandibular fracture. An incisional biopsy was performed, and the histopathologic examination demonstrated a malignant neoplasm of epithelial origin characterized by the proliferation of pleomorphic cells organized in sheets, nests, and cords and were also dispersed in a fibrous connective tissue stroma. Malignant neoplasms were discarded in other sites, and the diagnosis of PISCC was established. The patient underwent complete surgical resection of the lesion and is currently being followed up after 6 months without clinical-radiographic signs of relapse.

PRIMARY INTRAOSSEOUS SQUAMOUS CELL CARCINOMA: A REPORT OF 2 CASES

Primary intraosseous squamous cell carcinoma (PISCC) is a rare malignant neoplasm with a predilection for the mandible caused by remnants of the odontogenic epithelium or odontogenic cysts and tumors. We report 2 cases of PISCC and discuss its clinicopathologic features. Two female patients received diagnosis in an oral medicine service. The first one, 40-years-old, showed manifestation in the posterior mandible, pathologic fracture, and pain. In the second one, 78-years-old, the lesion was in a mental region with no symptoms and a reddish skin appearance in the chin region. The patients’ imaging examinations showed radiolucent lesions and bone cortical expansion. Both microscopic examinations revealed neoplastic epithelial cells arranged in cords and sheets exhibiting pleomorphism and cellular atypia. The connective tissue stroma was permeated by a moderate mononuclear inflammatory infiltrate. Both patients were surgically treated and are under follow-up.

Primary intraosseous squamous cell carcinoma of the mandible: report of two deceptive cases and literature review

Carcinoma as a primary tumor within the jaw is a rare phenomenon which usually arises from the epithelial lining of oral cavity or salivary glands. Primary intraosseous squamous cell carcinoma is a malignant neoplasm with no initial connection to oral mucosa. In this article, we reported two rare cases of primary intra-mandibular squamous cell carcinoma with clinical, radiographic, and histopathologic features and reviewed the literature.

Primary Intraosseous Squamous Cell Carcinoma of the Anterior Mandible Arising in an Odontogenic Cyst in 34-Year-Old Male

Primary intraosseous squamous cell carcinoma (PIOSCC) is a rare and aggressive malignancy arising exclusively within the jaws, without any initial connection with the oral mucosa. The etiology and the epidemiology are unclear due to the rarity of the disease, and there is no current universally accepted staging or treatment protocol. Clinically, the posterior mandible is the most affected site, and common symptoms are swelling and pain. The diagnosis is often difficult either because it requires stringent criteria to be satisfied or because of the absence of a pathognomonic histological pattern. Aggressive surgery is the first-choice treatment, often followed by radiotherapy. The lymph nodal status seems to be the most important factor influencing the prognosis, which is usually poor, with a 5-year survival rate ranging from 30% to 40%. In the present article, we report an unusual case of cystogenic PIOSCC interesting the anterior mandible of a young 34-year-old male, which came to our attention after complaint about recurrent infective episodes affecting a dentigerous cyst (impacted lower canine) discovered ten years before. The age, site, and extension are uncommon. Extensive surgical treatment with fibula free flap reconstruction, adjuvant therapy, and salvage surgery was carried out. The patient was disease-free at a 31-month follow-up.

A LATE DIAGNOSIS OF A PRIMARY INTEROSSEOUS SQUAMOUS CELL CARCINOMA: A CASE REPORT

<h3>Background</h3> Primary intraosseous squamous cell carcinoma is a rare malignant central jaw tumor derived from odontogenic epithelial remnants. It predominantly affects the mandible, although both jawbones may be involved. <h3>Objective(s)</h3> The purpose of this article was to represent the clinical, radiologic, and microscopic characteristics of a late diagnosed primary interosseous squamous cell carcinoma (PIOSCC). This case adds to the very few cases of PIOSCC in the literature. <h3>Study Design</h3> This case report describes a 60-year-old man who was initially misdiagnosed with a periapical infection related to the right lower wisdom tooth, that is, an infected odontogenic cyst of the mandible. After 4 months, the patient presented to a private dental clinic with a large swelling at the right side of the mandible. <h3>Results</h3> Extraoral examination revealed a swelling with paresthesia in the right mandibular angle. A panoramic radiograph was requested and revealed a complete erosion of the right ramus with an extension of the lesion to the body of the mandible. Advanced imaging revealed a large, ill-defined, right facial soft tissue mass lesion measuring 8.8 × 4.5 × 5 cm. The microscopic examination revealed fibrofatty tissue infiltrated by a tumor mass formed of solid groups of large pleomorphic atypical squamous cells with high nuclear-to-cytoplasmic ratio and focal intracytoplasmic keratin formation. The diagnosis was PIOSCC. <h3>Discussion/Conclusions</h3> Radiologic and clinical characteristics of PIOSCCs are similar to those of benign odontogenic cysts and tumors. In certain cases, early-stage PIOSCC may mimic routine dental disorders, such as periapical and periodontal disease, which may lead to misdiagnosis or delayed diagnosis. Clinicians must be aware that PIOSCC may initially present as a routine dental disorder, and that unsuccessful treatment may be a sign of underlying malignancy.

Condylar invasion after postoperative chemotherapy for primary intraosseous squamous cell carcinoma of the mandible: a case report

Abstract Primary intraosseous squamous cell carcinoma (PIOSCC) is a rare malignant tumor that occurs predominantly in the jaw. Pathological evidence shows that PIOSCC originates from the residual odontogenic epithelium and a preexisting odontogenic cyst or tumor. Here, we report the case of a 63-year-old man with central squamous cell carcinoma of the mandible. On the basis of imaging and histopathology reports, the patient was diagnosed with PIOSCC of the jaw. Subsequently, he was treated with postoperative adjuvant chemotherapy. During a postoperative follow-up visit in the ninth month, mandibular computed tomography (CT) scanning and bone imaging revealed local recurrence and condylar invasion. Therefore, radiation along with chemotherapy was administered. This case study adds to the literature on PIOSCC and widens the understanding of its diagnosis, treatment, and prognosis.

Primary intra osseous squamous cell carcinoma (PIOSCC) and TNM staging: a case report

Primary intra osseous squamous cell carcinoma (PIOSCC) and TNM staging: a case report