Primary intraosseous odontogenic carcinoma arising in an odontogenic cyst or de novo: A clinicopathologic study of six new cases

Department of Diagnostic Sciences and Pathology, University of Maryland, Baltimore, MD, USA.
Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology (Impact Factor: 1.46). 03/2006; 101(2):194-200. DOI: 10.1016/j.tripleo.2005.03.037
Source: PubMed


The term primary intraosseous odontogenic carcinoma (PIOC) has been primarily used to describe a squamous cell carcinoma within the jaws arising either from a previous odontogenic cyst or de novo. Here, we report 6 new cases of PIOC, affecting 4 female and 2 male patients with a mean age of 56.2 years. Two cases involved the maxilla and 4 cases occurred in the mandible. The typical radiographic presentation was that of a radiolucent lesion with well or ill defined margins. Histopathologically, 4 cases were diagnosed as well differentiated keratinizing PIOC arising from previous odontogenic cysts (2 odontogenic keratocysts and 2 periapical cysts). The remaining 2 cases were poorly differentiated nonkeratinizing PIOC, which appeared to arise de novo. Treatment consisted of surgical removal, with postoperative radiotherapy in 5 cases, and to date neither recurrence nor metastasis have occurred. Knowledge of the clinical, radiographic, and histopathologic features of PIOC allows accurate diagnosis and appropriate treatment of this rare malignancy.

19 Reads
  • Source
    • "PIOC has most frequently been found in the molar-ramus region of the mandible (15,16). The recurrent clinical symptoms are swelling, pain/toothache and lesion growth. "
    [Show abstract] [Hide abstract]
    ABSTRACT: Cyst‑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. A 59‑year‑old female was referred to Asahi University Murakami Memorial Hospital (Gifu, Japan), with acute pain in the right molars. An initial examination revealed buccal swelling and paresthesia of the mental nerve. Following an intraoral examination, the oral mucosa was confirmed to be normal, however, percussion pain was experienced between the lower right first premolar and second molar. Panoramic radiography revealed a retained lower right wisdom tooth and an irregular radiolucent area between the lower right molar and a mandibular angle with unclear margins. Computed tomography revealed diffuse bone resorption and an extensive loss of cortical bone on the buccal and lingual sides. A biopsy was performed and the pathological diagnosis was of a squamous cell carcinoma arising from the epithelial lining of the odontogenic cyst. Radical dissection was subsequently performed, however, histopathological examination of the resected specimen revealed neither invasion into the surrounding tissues penetrating the periosteum nor lymph node metastasis at the right submandibular lesion. Following the pathological diagnosis of primary intraosseous carcinoma (PIOC), the patient received 6,000 Gy radiation as post‑operative radiotherapy and chemotherapy with oral administration of tegafur, gimeracil and oteracil potassium. The patient is currently undergoing follow‑up examinations. Although PIOC arising from an odontogenic cyst is rare, it should be considered as a differential diagnosis for radiolucency of the jaw bone, particularly in older patients exhibiting a history of cystic lesions.
    Full-text · Article · Jun 2014 · Oncology letters
  • Source
    • "Unfortunately, the removed tooth and its associated tissues were not sent for histopathological examination, and this is still not uncommon in most parts of the world. Various odontogenic cysts have been associated with PIOSCC, including residual cyst, dentigerous cyst, odontogenic keratocyst, calcifying odontogenic cyst, and lateral periodontal cyst [5]. In analysis of 116 reported cases of PIOSCC arising in odontogenic cysts, the type of cyst more observed was residual/radicular with 70 cases followed by dentigerous cyst with 19 cases, keratocystic odontogenic tumor with 16 cases, 1 case of lateral periodontal, and 9 unclassified cases [8]. "
    [Show abstract] [Hide abstract]
    ABSTRACT: Primary intraosseous carcinoma of the jaws (PIOSCC) might arise from odontogenic epithelium, more commonly from a previous odontogenic cyst. The aim of this case is to illustrate that the clinician should consider that an apparent benign dentigerous cyst can suffer malignant transformation and that all material removed from a patient must be evaluated histologically. A 44-year-old man presented in a routine periapical X-ray an impacted lower left third molar with radiolucency over its crown. Ten years later, the patient complained of pain in the same region and the tooth was extracted. After one month, the patient still complained of pain and suffered a fracture of the mandible. A biopsy was performed and carcinoma was diagnosed. The patient was treated surgically with adjuvant radio- and chemotherapy and after 8 years, he is well without signs of recurrences. This report describes a central mandibular carcinoma probably developed from a previous dentigerous cyst.
    Full-text · Article · Apr 2014
  • Source
    • "The absence of a mucous cell component in PIOSCC, verified by a negative mucicarmine staining that we performed, serves to distinguish it from intraosseous mucoepidermoid carcinoma [29]. "
    [Show abstract] [Hide abstract]
    ABSTRACT: Primary intraosseous squamous cell carcinoma (PIOSCC) derived from an odontogenic keratocyst (OKC) is a rare malignant neoplasm of the jaws, which is locally aggressive with quite poor prognosis. The incidence of carcinomas arising in odontogenic cysts was reported to be approximately 1-2/1000. The number of well-documented cases of PIOSCC ex OKC is extremely small; hence, no sufficient incidence data are available in the literature. Overall, the survival rate of an individual, which is a period of two years, is very poor, and this can be attributed to the delayed diagnosis. But knowledge of the histopathological and immunohistological features of PIOSCC allows accurate and early diagnosis of the lesion so that an early and appropriate treatment can be instituted for better prognosis. The following report describes an extremely rare case of PIOSCC of the mandible derived from an OKC in a 20-year-old female patient.
    Full-text · Article · May 2013 · ecancermedicalscience
Show more