Clinicopathological Study of Primary Intraosseous Squamous Cell Carcinoma of the Jaw and a Review of the Literature

doi:10.1016/j.joms.2016.05.006

Journal of Oral and Maxillofacial Surgery

Volume 74, Issue 12, December 2016, Pages 2420-2427

https://doi.org/10.1016/j.joms.2016.05.006 Get rights and content

Purpose

Primary intraosseous squamous cell carcinoma (PIOSCC) is a rare malignant odontogenic tumor that originates from odontogenic epithelial remnants. It is often difficult to diagnose PIOSCC definitively; hence, extraction or surgical treatment is performed before the initial diagnosis in most cases. The present study examined new insights into and prognostic factors of patients with PIOSCC admitted to the authors' department.

Materials and Methods

An extensive record review was conducted of patients who underwent radical surgery for PIOSCC from January 2001 through December 2014.

Results

Of all cases of OSCC, the frequency of PIOSCC was 1.45%. The 2-year relapse-free survival (RFS) and overall survival (OS) rates were 50.0 and 41.6% in all cases, respectively. Three patients underwent surgery or tooth extraction before the initial diagnosis; in fact, intervention before initial diagnosis was found to be an important poor prognostic factor for RFS and OS. In contrast, patients who were not treated before the initial diagnosis was made did not exhibit any locoregional recurrence.

Conclusions

The treatment of PIOSCC should be similar to that for oral cancer with at least clinical stage T3N0 in the National Comprehensive Cancer Network clinical practice guidelines. In addition, cases of PIOSCC that are not treated before the initial diagnosis are more likely to obtain a good prognosis.

Section snippets

Patients

The authors retrospectively reviewed the records of patients who underwent radical surgery for PIOSCC from January 2001 through December 2014 and those who were followed for longer than 1 year. This study was approved by the independent ethics committee of the authors' hospital. Tumor stage was classified according to the TNM classification of the International Union Against Cancer,¹⁰ and histologic tumor differentiation was defined according to the WHO classification.

All patients underwent

Patient Characteristics

The clinicopathologic characteristics of the patients are presented in Table 1. During the 11-year period from January 2001 through December 2014, 6 of 414 patients (1.45%) were diagnosed with PIOSCC (5 men and 1 woman). The mean patient age was 71 years (range, 59 to 81 yr). The chief complaints included nonhealing of the extracted socket, pain and swelling, or swelling with sensory disturbance of the region. Three patients underwent surgery or tooth extraction before the initial diagnosis,

Discussion

The WHO 2005 report and the NCCN clinical practice guidelines have not provided detailed information regarding PIOSCC; hence, further insights and data on prognostic factors could help better elucidate the condition and develop novel treatments. In the present study, the authors aimed to elucidate new insights and the prognostic factors of PIOSCC.

PIOSCC is a rare malignant odontogenic tumor that accounts for fewer than 2% of all cases of OSCC.12, 13 The most common site of PIOSCC is from the

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This study aimed to explore the characteristics and prognoses of primary jaw malignancies and identify valuable prognostic factors.
Patients who were primarily diagnosed with jaw malignancies at Beijing Stomatological Hospital between 2006 and 2016 were retrospectively reviewed.
Eighty patients with malignant jaw tumors were enrolled in the study. Squamous cell carcinoma was the most common type (56.2%). The age range of patients varied from 11 to 92 years old (mean age = 51.5 years), and the male-to-female ratio was 2.1:1. Anatomically, the mandible was involved in 82.5% of cases. The estimated disease-free survival and overall survival (OS) rates were 60.6% and 73.2%, respectively. Cox multivariate analysis showed that a smoking history (hazard ratio = 5.015, 95% confidence interval, 1.900-13.235, P = .001) and advanced age (≥60 years; hazard ratio = 2.464, 95% confidence interval, 0.987-6.155, P = .046) were independent prognostic factors for poor OS.
Primary jaw malignancies were more prevalent in the mandible and among males and middle-aged patients. Radical resection of the primary surgery was the basis of a good prognosis, and the choice of postoperative adjuvant therapy may not ultimately affect OS. Patients with a history of smoking and advanced age were at high risk for poor prognosis.
Malignant Lesions Mimicking Endodontic Pathoses Lesion: A Systematic Review
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The prognosis for patients with PIOSSC is difficult to determine because of the small number of reported cases, the different treatment modalities, and the variable follow-up time. The overall survival rate of patients with PIOSCC was 40%–68% and 30%–40% at 2 and 5 years, respectively37. In addition, clear cell odontogenic carcinoma, a rare intraosseous tumor of the jaws, was 1 of the 6 carcinomas found.
Malignant nonendodontic periapical lesions represent a rare diagnosis, in which few case have been reported about this condition. Due to the low occurrence and little exploration of this subject, serious errors of diagnosis are observed in the oral medicine services. In this sense, the purpose of the present review was to integrate the information on the record published on malignant lesions that mimic endodontic pathoses into a comprehensive analysis of their clinical, radiologic, and histopathologic features.
An online search was made in March 2020 in the following databases: Pubmed, Web of Science, and Scopus. Eligibility criteria included publications having enough clinical, radiologic, and histopathologic information to confirm the diagnosis. Data were assessed descriptively.
Forty-nine publications from 16 countries comprising 60 cases were included. There is a nearly equal occurrence rate between males and females (male/female ratio is 49.15%/50.84%) with a mean age of 46.56 years. The lesions were more prevalent in the posterior mandibles (40.69%), and, radiographically, they were unilocular radiolucent. Histopathologic analysis showed metastasis (26.67%) as the most prevalent lesion followed by salivary gland malignant disease (25%).
The present systematic review revealed several histologic types of malignant periapical lesions that could be mimicking endodontic pathoses. Because these lesions usually do not show clinical or radiologic features of malignant alteration, clinicians should consider these hypotheses in their routine.
Remarkable Short-Term Regression of a Posterior Mediastinum Metastasis From Primary Intraosseous Carcinoma Treated With Nivolumab: A Case Report
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Thus, further investigation is required to clarify this correlation because of the rarity of this tumor. Although the NCCN clinical practice guidelines have not described any standard therapy for PIOC,3 wide surgical resection is currently the gold standard.2,12 Several reports have suggested that postoperative radiotherapy could improve overall survival.
Primary intraosseous carcinoma (PIOC; not otherwise specified) is a rare odontogenic malignancy that is believed to arise from the odontogenic epithelium. In addition, some cases arise in odontogenic cysts or other benign precursors. There are no established treatment protocols for PIOC because of its rarity. In particular, no standard therapy has been established for patients with recurrent or metastatic PIOC. This report describes a case in which the use of nivolumab showed a marked treatment response in metastatic PIOC. The patient was a 71-year-old man with PIOC. The patient underwent segmental mandibulectomy with modified radical neck dissection after neoadjuvant chemotherapy, including 2 cycles of paclitaxel, cetuximab, and carboplatin and 1 cycle of docetaxel, cisplatin, and 5-fluorouracil. The lesion rapidly enlarged during neoadjuvant chemotherapy, and surgical specimens showed no histologic therapeutic effects of chemotherapy. Three months after the surgery, contrast-enhanced computed tomograms showed posterior mediastinal lymph node metastasis. Treatment with nivolumab was initiated, and notable clinical improvement was noted after 2 cycles. Although it was a short-term treatment, remarkable effects were observed and no adverse events were noted during and after nivolumab treatment. Nivolumab could be an attractive treatment option for metastatic PIOC that is resistant to traditional chemotherapy and target therapy, including cetuximab. Further investigation of this treatment is warranted.
Risk factors and prognosis for the primary intraosseous carcinoma of the jaw
2019, International Journal of Oral and Maxillofacial Surgery
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In general, the overall prognosis for PIOC is poor with low survival rates and high recurrence rates. Previous studies reported that the 2-year survival rate was around 60∼70%, while 5-year survival rate was around 30∼40%15,16,19. Our study showed similar estimated results.
Primary intraosseous carcinoma (PIOC) is a rare but aggressive type of odontogenic tumour arising within the jawbone. Diagnosis criteria and treatment strategy remain difficult and controversial. The present study aimed to clarify the clinicopathological features and determine prognostic factors in management of PIOC. A retrospective study of 30 patients with PIOC, treated at the Hospital of Stomatology of Sun Yat-sen University between 2009 and 2017, was conducted. Clinical, histopathological and treatment modality data were collected. Follow-up data were recorded to determine prognostic factors. There were 19 males and 11 females with a mean age of 52.3 years. The most common location of the tumour was the mandible (90%). Having a history of tooth extraction or tooth mobility was the major characteristic symptom (63.3%), jaw swelling coming in second (53.3%). Half of the patients underwent surgery alone. The estimated 2-year overall survival rate (OS) and recurrence-free survival rate (RFS) were 61.3% and 40.1%, respectively. Higher histological grade was an independent risk factor for poor OS (hazard ratio (HR) 0.233 [0.059–0.915], P = 0.037), while at pN₊ stage for RFS, HR = 5.627 [1.199–26.409], P = 0.029. Because of its rarity and intrabony site, the classification, staging and treatment guidelines for PIOC should be further studied and established.
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Primary intraosseous squamous cell carcinoma derived from a keratocystic odontogenic tumor with a large inflammatory component. Case report and a review of the literature
2017, Revista Espanola de Patologia
El desarrollo de un carcinoma de células escamosas a partir del revestimiento epitelial de un tumor odontogénico queratósico (TOQ) corresponde a una neoplasia maligna infrecuente exclusiva de los huesos maxilares, denominada también cómo carcinoma de células escamosas intraóseo primario (CCEIP). Afecta principalmente a personas de mediana edad, especialmente a hombres (2:1) y se localiza usualmente en la zona posterior de la mandíbula. Aunque la patogénesis del CCEIP es desconocida y no se le puede atribuir asociación con factores predisponentes para el desarrollo de carcinoma de células escamosas de la mucosa oral, actualmente se refuerza la idea del componente inflamatorio como predisponente de malignización. Se presenta un caso clinicopatológico de un varón de 61 años de edad con diagnóstico de CCEIP derivado de un TOQ con un gran componente inflamatorio asociado.
The development of a squamous cell carcinoma from the epithelial lining of a keratotic odontogenic tumor (KOT) corresponds to a rare malignant neoplasm exclusive to the jawbone, also referred to as primary intraosseous squamous cell carcinoma (PIOC). It mainly affects middle-aged people, especially men (2: 1) and is usually located in the rear area of the jaw. The pathogenesis of PIOC is unknown and no predisposing factor for the development of squamous cell carcinoma of the oral mucosa has been identified. However, many authors support the hypothesis that a component of a chronic inflammatory process could prove to be the underlying factor of malignant change. We present a case of a 61-year-old male diagnosed with PIOC derived from a KOT associated with an extensive inflammatory process.

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PathologyClinicopathological Study of Primary Intraosseous Squamous Cell Carcinoma of the Jaw and a Review of the Literature

Purpose

Materials and Methods

Results

Conclusions

Section snippets

Patients

Patient Characteristics

Discussion

Oral Oncol

Oral Surg

J Oral Maxillofac Surg

Int J Oral Maxillofac Surg

J Oral Maxillofac Surg

Oral Surg Oral Med Oral Pathol Oral Radiol Endod

J Craniomaxillofac Surg

J Oral Maxillofac Surg

Oral Surg Oral Med Oral Pathol Oral Radiol

Oral Surg Oral Med Oral Pathol Oral Radiol

Br J Oral Maxillofac Surg

Pathology
Clinicopathological Study of Primary Intraosseous Squamous Cell Carcinoma of the Jaw and a Review of the Literature