Primary intraosseous carcinoma arising from an odontogenic cyst: a case report and review of the Japanese cases

A rare case of primary intraosseous carcinoma (PIOC) arising from an odontogenic cyst in a 58-year-old man is reported. Clinical and radiological examinations revealed an odontogenic cyst of the maxilla. Histopathologically, the lesion was composed of a cyst with a parakeratotic epithelial lining and well-differentiated squamous cell carcinoma, showing continuity between them without a connection to the oral mucosa. Twenty-eight well-documented Japanese cases of Type-1 PIOC, including the present case, were reviewed. The mean age of the 28 patients was 56.1 years, and the male to female ratio was 1.8:1.0. Compared with currently reported Japanese reviews of Type-3, foreign Type-1 and Type-3, there were no significant differences in mean patient age and sexual predominance, and no racial difference. The pathogenesis of Type-1 PIOC is also discussed.     

Primary de novo intraosseous carcinoma of the mandible--report of a case and review of literature.

Primary intraosseous carcinoma (PIOC) is a rare malignant odontogenic neoplasm defined as squamous cell carcinoma arising within thejaws, having no initial connection with the oral mucosa and presumably developing from the residues of the odontogenic eptihelium. A 37yr old male patient reported with the complaint of pain and swelling in the right mandible and inability to open his mouth for the past six months. Examination revealed an expansile mass that involved the right mandible and extended from the 1st premolar region to the right angle of the mandible. The alveolar ridge was markedly expanded and was covered with intact, normal appearing mucosa. Radiographic examination revealed an ill-defined radiolucent area in the right body of the mandible, which extended to the lower border. Histopathologic examination revealed a well differentiated keratinizing squamous cell carcinoma. PIOC has no specific histopathologic features and can be definitively diagnosed only upon fulfillment of various criteria. These criteria were followed in our case and reported here for its rarity.     

原发性颌骨内鳞状细胞癌的诊断与治疗

目的:回顾性研究分析临床少见的原发性颌骨内鳞状细胞癌(PIOC)的临床病理表现,为选择合理的治疗方案提供帮助.方法:对1995-02--2007-01间收治的经病理证实的12例PIOC的临床表现、影像学特征、治疗特点等进行综合分析.结果:12例PIOC患者中以中老年多见,平均年龄45.2岁,全发生于下颌骨;临床特点以下...     

Primary intraosseous carcinoma associated with impacted third molar of the mandible: review of the literature and report of a new case

This study investigated the clincopathologic features of primary intraosseous carcinoma (PIOC) related to the third molar of the mandible. Six cases (5 from the literature and 1 new case; mean patient age 73 years) of PIOC associated with completely impacted lower third molar were analyzed. For comparison, 8 cases of PIOC presenting after extraction of the lower third molar were also evaluated, with a significantly lower mean patient age of 56 years. Most lesions were deceptively similar to those related to the lateral type of dentigerous cyst, and half of the impacted cases revealed an asymptomatic fracture of the mandible. In all patients except for the present case, metastasis had not been recorded. Surprisingly, no patients are known to have died of PIOC. In our experience the prognosis of PIOC associated with a completely impacted third molar is much better than commonly believed, although the number of cases involved is too small to make a general conclusion. However, delay in establishing the correct diagnosis followed by extraction without awareness of the carcinoma is know to adversely affect the prognosis.     

Intraosseous carcinoma of the anterior maxilla identified as the occult primary tumour of carcinoma of unknown primary

Carcinoma of unknown primary (CUP) is defined as lymph node metastasis without a detectable origin until after the initial treatment has been performed. The most common occult primary site in the head and neck, as revealed by a review of the published literature, is the oropharynx. An occult primary site in the oral region is extremely rare. We report a rare case of head and neck CUP (HNCUP) in a 69-year-old female patient, wherein the occult primary lesion was a primary intraosseous carcinoma (PIOC) invading the anterior maxilla. During the course of the initial diagnostic workup, no primary lesion could be identified; however, cervical lymph node metastasis to left levels IB and IIA were observed in the patient. A neck dissection followed by adjuvant radiotherapy was performed. However, the PIOC of the anterior maxilla was identified 6 months after neck treatment and was confirmed as the occult primary tumour of the HNCUP. This case is quite rare and required a comprehensive workup to guide optimal treatment. Careful follow-up or active biopsy should be considered if osteolytic changes are observed in the jaw.     

Primary intraosseous carcinoma of the mandible

The case of a 58-year-old man with a large primary intraosseous carcinoma (PIOC) of the mandible is presented. The tumour is rare, and the prognosis is usually poor. There are no specific histological characteristics of PIOC. Staining for basement membrane laminin could be a valuable aid in the differential diagnosis between PIOC and tumours of ameloblastoma origin.     

原发性颌骨内癌14例临床分析

目的：探讨原发性颌骨内癌的治疗与预后。方法：对1999—2009年收治于中国医学科学院肿瘤医院的14例原发性颌骨内癌患者进行回顾性分析。采用SPSS13.0软件包对资料进行统计学分析。结果：14例患者中,12例行综合治疗,2例行单纯手术治疗。患者1、2、3年的累计生存率分别为85.1%、46.4%和23.2%,而患者1,2,3年的累计无复发生存率分别为50.0%、35.7%和17.9%。Logistic多因素回归分析显示,年龄、性别、病理分级、淋巴结转移及治疗方式均不是患者预后的独立影响因素。结论：根治性手术切除是原发性颌骨内癌的主要治疗方式,早期治疗相当重要;对于切除不彻底的患者,可考虑辅助放化疗,但疗效有待证实。     

Primary intraosseous carcinoma of the mandible with probable origin in an odontogenic cyst

Primary intraosseous carcinoma of the jaws (PIOC) is an uncommon lesion, but may not be as rare as commonly believed. Since the putative source of the epithelium giving rise to an intraosseous carcinoma is the epithelium involved in odontogenesis, these lesions are often designated as odontogenic carcinomas. These tumors may theoretically arise (1) from the lining of odontogenic cysts, (2) from other epithelial odontogenic tumors, or (3) de novo from presumed odontogenic rests. While not included in most classifications of PIOC, it appears logical to also include intraosseous mucoepidermoid carcinomas as a fourth type of PIOC. A case of primary intraosseous squamous cell carcinoma of the mandible, with evidence of origin in an odontogenic cyst, is presented. The recent literature on carcinomas arising in jaw cysts is reviewed.     

Risk factors and prognosis for the primary intraosseous carcinoma of the jaw

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.     

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

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.     

腮腺皮脂腺癌1例

本文报告1例腮腺皮脂腺癌。患者男67岁,右侧腮腺区无痛性肿物1年。专科检查右腮腺区触及一大小约2 cm×2 cm×1.5 cm肿物。CT检查右侧腮腺浅部类圆形肿块影,边界模糊。病理镜下见透亮细胞、泡沫样细胞呈巢状、腺样或片状排列,免疫组化示:ACTIN(-)、CK(+)、GFAP(-)、P63(+)、S-100(-)、Vinmentin(-)。诊断为腮腺皮脂腺癌。     

Squamous cell carcinoma arising in a residual cyst: A case report

A case of squamous cell carcinoma (SCC) arising in a mandibular residual cyst in a 62-year-old man is presented. The treatment included enucleation followed by primary closure. Histologic examination revealed a poorly differentiated SCC in the cyst lining without invasion through the connective tissue wall. Eight and a half years later, the patient was still free from recurrence.     

Primary intraosseous carcinomas of the jaws arising within an odontogenic cyst, ameloblastoma, and de novo: report of new cases with reconstruction considerations

Primary intraosseous carcinoma (PIOC) of the jaws has been rarely reported. The authors report 3 new cases of PIOC arising within an odontogenic cyst, ameloblastoma, and de novo origin, respectively. Surgeons should appreciate the elevated aggressiveness of this tumor despite adequate surgical treatment. The authors recommend initial aggressive surgical treatment to decrease the local recurrence rate.     

Trichilemmal carcinoma of the scalp

This report describes the case of an 86-year-old male who presented with a large scalp tumour. Imaging revealed a large vertex mass, without intracranial extension, and multiple nodular subcapsular hepatic lesions suspected to be secondary in nature. Surgical resection was performed. Pathological examination revealed an adnexal carcinoma of follicular origin, thus a trichilemmal carcinoma. Controlled wound healing (budding of the diploë) was completely successful within 12 months. The patient refused the assessment and treatment of his metastases.     

成釉细胞癌的临床及病理特点：2例报告及文献复习

目的: 分析总结成釉细胞癌的临床及病理特点。方法：分析2例成釉细胞癌患者的临床资料,并检索和回顾分析相关中、英文文献。结果：收集临床资料完整的文献30篇, 包括成组病例分析3篇, 个案报道27篇, 共计61例。结论：成釉细胞癌是一种罕见的牙源性上皮性恶性肿瘤,无明显性别差异,缺乏特异性临床表现,明确诊断需依据病理和免疫组化。其侵袭性强,首选手术治疗,预后一般,需长期随访。     

Primary intraosseous carcinoma of the jaw: pooled analysis of world literature and report of two new cases

Primary intraosseous carcinoma (PIOC) is a rare malignant neoplasm of the jaw. These tumours are believed to arise from the odontogenic epithelium and hence are also referred to as odontogenic carcinoma. A detailed search was made for squamous cell PIOC of the jaw in English literature using Medline Cancer CD. The data obtained were transferred onto dBase software. Two detailed case reports of patients treated at Regional Cancer Centre, Trivandrum during 1996 and 1997 were also included. A pooled analysis was carried out. Survival analysis was carried out using Kaplan-Meier method and log-rank statistics were used for comparing survival. A total of 35 cases were analysed, of which 33 were from published literature. The mean age of the patients at the time of diagnosis was 52.3 years with male to female ratio being 2.5:1. Posterior mandible was the predominant site. The median follow-up time was 28 months. Overall survival at 5 years was 37.8% (95% CI; 14.8-61.0) while the corresponding disease free survival was 29.8% (95% CI; 9.2-54.1). Primary intraosseous carcinoma is a rare tumour of jawbones, characterized by progressive swelling of the jaw, pain and loosening of tooth. The tumour is locally aggressive and metastasizes to regional nodes. The overall and disease free survival is poor with almost 50% patients failing loco-regionally within the first 2 years of follow-up.     

Central acinic cell carcinoma of the mandible. Case report

Central acinic cell carcinoma (of the mandible) is rare, and, to our knowledge, only seven cases of this disease have been reported in the literature. A case in a 67-year-old Japanese woman is presented. Clinical examination revealed a 10.0x6.0mm mass located on the buccal aspect of the gingiva of the second molar in the left mandible. Radiographic examination revealed a radiolucency from the second to the third molar of the left mandible. Computed tomography disclosed destruction of the lingual cortical bone of the third molar region. The preliminary diagnosis was of odontogenic tumour. The patient was admitted, and removal of the tumour and of the involved teeth were carried out. Histological examination disclosed the diagnosis of acinic cell carcinoma. Subsequently, the tumour area was widely excised from the second premolar region to the coronoid process, and radical neck dissection was performed. A lymph node metastasis was found in the submandibular region. No recurrence or metastasis was observed during the 34-month follow-up.     

Hepatocellular Carcinoma Metastatic to the Gingiva as a First Manifestation of Hepatocellular Carcinoma

A 55-year-old man consulted to our hospital complaining of a gingival tumor. Oral examination showed granulation tissue-like polypoid tumors (2 × 2 × 1 cm) in the left upper gingiva. A clinical diagnosis of granulation tissue was made, and an incisional biopsy was performed. It showed carcinoma cells in the submucosa. They were arranged in compact and sinusoidal patterns. A few bile plugs and one Mallory-like body were recognized by meticulous examination. An immunohistochemical study showed that the tumor cells were positive for hepatocyte paraffin-1 (HepPar-1), cytokeratins (AE1/3 and CAM6.2), and p53 protein. The carcinoma cells were negative for vimentin, prostate specific antigen, CD10, and α-fetoprotein. The Ki-67 labeling was 49%. A histological diagnosis of hepatocellular carcinoma (HCC) metastatic to the gingiva was made. Then, the patient was referred to hepatology unit. A blood laboratory test showed elevated elevated liver enzymes, and positive antibody to hepatitis C virus. Liver cirrhosis was also present. Imaging modalities including US, CT, and MRI revealed multiple hepatic tumors. Metastases to other sides were not recognized. The patient was diagnosed as cirrhosis and HCC with an extrahepatic metastasis to the gingiva. The patient was now treated by chemotherapy. The present case shows that HCC can manifest as small gingival tumors, and meticulous HE examination and immunoreactive HepPar-1 are useful in the diagnosis of HCC in extrahepatic metastatic sites.     

基底细胞痣综合征手术治疗同期前臂皮瓣转移修复1例报告

本文报告1例男性基底细胞痣综合征患者,30岁,全麻下行颌骨多发囊肿切除术、下颌软组织及骨组织扩大切除术,同期使用前臂游离皮瓣移植,修复下颌皮肤及口腔黏膜缺损,术后恢复良好。     

应用快速冰冻活检确定唇癌手术边缘的研究

目的：评价快速冰冻活检在唇癌手术治疗中的意义。方法：分析168例经手术治疗的唇癌患者术中快速冰冻活检确认手术边缘的检查结果。手术边缘发现浸润癌或原位癌记为阳性边缘。结果：168例患者中有22例发现阳性边缘（13．10％）,168例中10例（5．95％）术后复发。结论：快速冰冻活检评估手术边缘是控制手术切除范围的可靠方法,可有效减少术后复发及术后放疗,但阴性手术边缘并不能保证术后的零复发率。