Plasmacytoid myoepithelioma of a minor salivary gland

Plasmacytoid myoepitheliomas are rare salivary gland neoplasms that probably represent a variant of the pleomorphic adenoma. They appear to occur more frequently in the minor salivary glands of the mouth. The mean average age at time of diagnosis of intraoral lesions is 18.8 years. Cytologic pleomorphism is a frequent histologic feature, which has led to a questionable diagnosis of malignancy in five out of ten cases. Caution is advocated in the evaluation of a salivary gland neoplasm with such features, and confusion with primary squamous cell or undifferentiated carcinoma of the salivary glands should be avoided. Patients who have cytologically pleomorphic lesions should be assessed for evidence of metastases. Rapid growth, neurologic evidence of sensory or motor dysfunction, and bone invasion should be regarded as ominous clinical parameters. Therapy should be directed toward complete surgical extirpation.     

颌下腺转移性多形性腺瘤1例

本文报道1例罕见的转移性多形性腺瘤,患者女性,30岁,左颌下腺及右锁骨上包块术后病检结果与患者8年前左颌下腺肿块切除后病检结果一致,病理形态均表现为良性多形性腺瘤。并结合文献报道,对转移性多形性腺瘤的临床病理特征、发病机制及防治方法等进行讨论。     

1878例涎腺肿瘤WHO组织病理学新分类的统计分析

目的 根据ＷＨＯ１９９０年涎腺肿瘤组织病理学新分类标准对１８７８例涎腺肿瘤及肿瘤样疾病进行重新分类,并重点对涎腺上皮性肿瘤的临床发病情况进行统计分析。方法 统计描述。结果 １８７８例涎腺肿瘤以诞腺上皮性肿瘤为主,为１４３１例,占７６．２０％,其中腺瘤发病居首位,其次是涎腺癌,涎腺肿瘤样疾病居第三位;多形性腺瘤是涎腺肿瘤中最常见者,恶性多形性腺瘤为最常见的涎腺癌,特别注意了腺癌亚分类中的几种少见类型     

Enucleation of a pleomorphic adenoma of the palate: a conservative choice

The benign mixed tumor (pleomorphic adenoma) is the most common intraoral salivary gland tumor: more than 50% occurs on the hard and soft palate. The epidemiological data, histopathological and clinical features, preoperative diagnostic methodologies, treatment options, recurrence rates and prediction published in the literature for this neoplasm are discussed. A case of minor salivary gland tumor of the palate in a 26 year old man is reported. The therapy chosen was a surgical conservative enucleation of the lesion. Histological examination showed pleomorphic adenoma; there is no recurrence after 2 years.     

下颌下腺非侵袭性恶性多形性腺瘤1例报告

唾液腺非侵袭性恶性多形性腺瘤(Ca-ex-PA)又称发生在多形性腺瘤中的原位癌,是较少见的病理类型,临床表现与良性多形性腺瘤相似,有条件完整切除肿瘤,复发或转移少见,预后良好.本文报告1例下颌下腺非侵袭性多形性腺瘤的临床病理学特征,认为鉴别非侵袭性Ca-ex-PA和侵袭性Ca-ex-PA对判断预后十分重要,并影响治疗方案的选择,WHO对唾液腺多形性腺瘤恶变的分型在肿瘤的生物学行为和预后方面具有实际意义.     

Maintaining a high level of suspicion for recurrent malignant disease: report of a case with periapical involvement

Aim: To report the unusual endodontic presentation and radiographic features of a subgroup of pleomorphic adenoma called carcinoma ex pleomorphic adenoma and to stress the importance of maintaining a high level of suspicion in cases where primary or recurrent neoplasia is included in the differential diagnosis. SUMMARY: This paper describes a case in which a patient with a previous history of malignant neoplasm presented with signs and symptoms similar to a dental infection. The pathology report however confirmed recurrence of the previous tumour in a malignant fashion. The importance of maintaining a high level of suspicion in early management of such disease is highlighted. KEY LEARNING POINTS: *Pleomorphic adenomas, also known as benign mixed tumours, are common salivary gland tumours, which infrequently undergo malignant transformation, with potentially devastating consequences. *Malignant salivary gland tumours can present as dental swelling, dental pain and sudden loss of vitality of teeth so both general practitioners and specialists have the responsibility to evaluate such patients with a broad vision. *Radiographic differential diagnosis of periapical radiolucency should also include malignant salivary gland tumours. *This case highlights the need of vigilance at all times and emphasizes the benefits of biopsy and histological examination in the diagnosis of recurrent malignant salivary gland tumours.     

基质金属蛋白酶在涎腺良、恶性多形性腺瘤中的表达

目的:检测基质金属蛋白酶2(MMP-2)、膜型基质金属蛋白酶1(MT1-MMP)、基质金属蛋白酶组织抑制剂2(TIMP-2)及细胞外基质金属蛋白酶诱导因子(EMMPRIN),在人正常涎腺组织和涎腺良、恶性多形性腺瘤中的表达,探讨其表达的生物学意义。方法:通过免疫组织化学技术SP法检测人66例正常涎腺组织、45例涎腺多形性腺瘤及42例涎腺恶性多形性腺瘤中,MMP-2、MT1-MMP、TIMP-2及EMMPRIN的表达,采用χ2检验比较3种组织中MMP-2、MT1-MMP、TIMP-2及EMMPRIN表达的差异。结果:MMP-2在人正常涎腺组织,涎腺良、恶性多形性腺瘤中的阳性表达率分别为9.09%、46.67%、85.71%;MT1-MMP在以上3种组织中的阳性表达率分别9.09%、53.33%、78.57%;TIMP-2在以上3种组织中的阳性表达率分别为10.61%、44.44%、59.52%;EMMPRIN在以上3种组织中的阳性表达率分别为13.64%、48.89%、83.33%。MMP-2、MT1-MMP、TIMP-2及EMMPRIN在涎腺多形性腺瘤中的表达显著高于人正常涎腺组织,且MMP-2、MT1-MMP及EMMPRIN在涎腺良、恶性多形性腺瘤中表达的差异有统计学意义。结论:在涎腺多形性腺瘤中,MT1-MMP、TIMP-2及EMMPRIN的表达与MMP-2的活化有关,且MMP-2、MT1-MMP、TIMP-2及EMMPRIN有可能作为判断多形性腺瘤侵袭性的有效指标。     

大涎腺上皮性肿瘤212例临床分析

目的 总结大涎腺上皮性肿瘤的临床特点,指导临床实践。方法 按照WHO 1991年涎腺肿瘤组织学分类标准,对212例经病理确诊的大涎腺上皮性肿瘤进行回顾性临床分析,讨论良、恶性肿瘤的不同临床特征。结果 212例肿瘤中,腺瘤166例(78.3％),以多形性腺瘤为主;涎腺癌46例(21.7％),以腺样囊性癌最多见。腮腺是最主要发病部位。涎腺腺瘤临床多表现为无痛性肿块,涎腺癌常伴有疼痛和(或)神经麻痹等症状。结论 大涎腺良、恶性肿瘤具有不同的临床特征,有助于临床正确诊断。     

Neural adhesion molecule (N-CAM) in pleomorphic adenoma and carcinoma ex-pleomorphic adenoma

BACKGROUND: The neural cell adhesion molecule (N-CAM) has been implicated in the behaviour of the adenoid cystic carcinoma. In vitro, it was demonstrated that N-CAM inhibits cell invasion. The aim of this study was to search for N-CAM in the most common salivary gland tumour that has a malignant counterpart. METHODS: We investigated the presence of N-CAM in pleomorphic adenoma and its malignant counterpart, the carcinoma ex-pleomorphic adenoma, using the immunohistochemistry technique. RESULTS: Neural cell adhesion molecule was expressed in all cases of pleomorphic adenoma, strongly labelling the luminal cells of the double-layered ductform structures. This expression was weaker in neoplastic myoepithelial cells and progressively diminished at a distance from the luminal cells. In carcinoma, ex-pleomorphic adenoma N-CAM was either totally absent or faintly present at the apical pole of the few luminal cells. CONCLUSIONS: As a result of the peculiar distribution of N-CAM in pleomorphic adenoma, we speculated that N-CAM behaves as a tumour-suppressor molecule, which is expressed in the benign neoplasm and which is down-regulated after malignancy, when the tumour assumes an invasive behaviour.     

涎腺多形性腺瘤恶变机制的研究进展

涎腺多形性腺瘤是最常见的涎腺上皮性肿瘤,多次复发或病程长者可恶变为恶性多形性腺瘤.涎腺多形性腺瘤的恶变是多因素、多基因变异的综合病变过程.研究发现,细胞遗传学改变、癌基因、抑癌基因、黏附分子及细胞外基质蛋白在涎腺多形性腺瘤的恶变过程中均有一定的作用.本文就多形性腺瘤恶变机制的研究进展作一综述.     

Pleomorphic Adenoma of Minor Salivary Gland in a 14 year Old Child

Salivary gland tumours are rare in childhood, and almost all of them occur in parotid gland. Minor salivary gland tumours are even rarer, pleomorphic adenoma being the most frequently found tumour. Only seventeen cases of pleomorphic adenoma arising in the minor salivary gland tumour have been reported in children and adolescents. Pleomorphic adenoma of minor salivary gland represents about 45% of all the tumours of the minor salivary glands. Pleomorphic adenoma is slowly enlarging tumour indistinguishable from adenoid cystic carcinoma clinically, except for pain and ulceration, which is more common in the latter. Carcinoma arising from pleomorphic adenoma has been reported in 3% cases amongst the minor salivary gland tumours. This report presents a case of pleomorphic adenoma of minor salivary gland in a 14 year old female patient with a brief review of literature.     

Synchronous mucoepidermoid carcinoma of tongue and pleomorphic adenoma of submandibular gland

Presentation with synchronous salivary gland tumors is rare, with the most typical combination being Warthin's tumor and pleomorphic adenoma of the parotid gland. Involvement of minor salivary glands in such occurrences is extremely uncommon. We report a case of simultaneous mucoepidermoid carcinoma of the tongue and pleomorphic adenoma of the submandibular gland in a 40-year-old woman. The submandibular mass was initially considered to represent regional lymph node tumor metastasis but later was shown to be an intraglandular primary neoplasm.     

涎腺肿瘤中Pax9基因的表达及其意义

目的:检测正常涎腺组织及涎腺肿瘤中转录因子Pax9的表达。方法:采用免疫组化SABC法,研究Pax9在正常涎腺组织、多形性腺瘤、腺淋巴瘤、基底细胞腺瘤、粘液表皮样癌和腺样囊性癌共48例中的表达情况。结果:除1例多形性腺瘤外,其余组织均表达Pax9。正常涎腺的腺泡细胞和导管内衬细胞、多形性腺瘤的上皮细胞、腺淋巴瘤的肿瘤上皮细胞和基底细胞腺瘤中Pax9弱阳性表达,差异没有显著性;而在增殖活跃的细胞如正常涎腺导管的基底细胞和恶性肿瘤细胞(粘液表皮样癌、腺样囊性癌)中表达增强,恶性肿瘤中Pax9的强阳性表达率明显增高,与正常和良性肿瘤相比具有显著差异(P<0.05)。结论:Pax9在涎腺肿瘤尤其是恶性肿瘤的发生过程中发挥重要的作用。     

Neck mass with marked squamous metaplasia: a diagnostic pitfall in aspiration cytology

Fine-needle aspiration cytology (FNAC) is a highly accurate tool for the diagnosis of pleomorphic adenomas; however, this common salivary gland neoplasm can be diagnostically challenging, causing pitfalls in cytodiagnosis. A 50-year-old woman suffered from a painless, slowly-growing mass in the right posterior upper neck for 5 months. FNAC from this mass revealed many discohesive parakeratotic cells and keratin flakes, and a few stromal elements. Under the impression of metastatic well-differentiated squamous cell carcinoma, the tumor mass was totally excised. Histology confirmed a pleomorphic adenoma with marked squamous metaplasia and frequent keratin pearl formations without the evidence of malignancy. Pleomorphic adenoma occasionally reveals focal squamous metaplastic changes, when extensive, it may be misdiagnosed as metastatic well-differentiated squamous cell carcinoma in FNAC, particularly in the neck region.     

Simultaneous palatal pleomorphic adenoma and mandibular keratocystic odontogenic tumor in a pediatric patient

BACKGROUND: Neoplasms of the salivary gland account for 3% of all head and neck tumors; most of these occur in the parotid gland and are benign; 80% of them are pleomorphic adenomas. Benign neoplasms of the salivary gland are rare in children compared with adults. Keratocystic odontogenic tumor (KCOT) is considered the second most frequent neoplasm of all odontogenic tumors after the ameloblastoma. In the present report, we introduce the unusual presentation of a child with a simultaneous pleomorphic adenoma of the right hard palate and a KCOT of the left mandibular angle. DISCUSSION: The case is significant for 2 reasons; according to the literature, there are few reports of pleomorphic adenoma arising from a minor salivary gland of the palate in a patient younger than 18 years. This is to our knowledge the first recorded instance of simultaneous palatal pleomorphic adenoma and mandibular KCOT occurring in a child.     

Carcinoma ex pleomorphic adenoma of the upper lip

Carcinoma ex pleomorphic adenoma (CXPA) is a rare salivary gland malignancy most often reported within the parotid gland. Of the salivary gland tumours that occur within the minor salivary glands at least 50% are reported to be malignant. This proves to be inaccurate when describing salivary gland tumours within the upper lip which are usually benign. A Medline search of the English language literature yields only one case report of a CXPA located within the upper lip. The authors present a second case report of CXPA within the upper lip and a review of its pathologic features and management.     

Epithelial salivary gland tumors of children and adolescents in southern Portugal. A clinicopathologic study of twenty-four cases.

During a 30-year period 24 epithelial salivary gland tumors were diagnosed in children and adolescents less than 18 years of age. The cases were retrieved from a series of 759 consecutive cases of salivary gland tumors (3.2%) from the area corresponding to southern Portugal during the same period of time. The mean age of the patients was 13.4 years, and one case was congenital. There was a slight female predominance (male/female ratio 1:1.7). The parotid gland was affected in most cases (70.8%). Seventeen neoplasms were benign, and the remaining seven were malignant. As in the adult group, pleomorphic adenoma was the most frequent benign tumor (66.6%), with similar histologic findings and clinical course. Mucoepidermoid carcinoma was the prevalent malignant tumor (20.8%), had a high grade of differentiation, and had a favorable outcome. The histologic pattern of the congenital neoplasm was similar to that of adult epithelial-myoepithelial carcinoma.     

Tumours of the salivary glands: a histological and clinical review.

The aim of this paper is to try to identify points of correlation between the histological appearance and the clinical behaviour of minor salivary gland tumours. To this end 62 well-documented minor salivary gland tumours have been followed up for at least five years or until recurrence. The discussion is limited to the adenoidcystic carcinoma, the pleomorphic adenoma and the basal cell adenoma, since only these three tumours were represented in sufficient numbers to be analysed. For each tumour a number of histological pointers to prognosis are reviewed. In the case of the basal cell adenoma, a recently recognised tumour, the possibility of confusion with the adenoidcystic carcinoma is discussed.     

基质金属蛋白酶及其组织抑制剂在涎腺多形性腺瘤生物学行为中的意义

目的 研究涎腺多形性腺瘤中基质金属蛋白酶(MMP)-2、9及其组织抑制剂(TIMP)-1、2的表达变化与其生物学行为的关系。方法 用免疫组化SP法和明胶酶谱法分别检测9例普通型和14例生长活跃型多形性腺瘤中MMP-2、9及TIMP-1、2的阳性表达及细胞定位，分析其中酶原与活性酶的含量比例。结果 MMP-2及MMP-2／TIMP-1、MMP-2／TIMP-2的比值在生长活跃型多形性腺瘤中的表达高于普通型，活性MMP-2、MMP-9酶原和活性酶在生长活跃型多形性腺瘤中的表达明显高于普通型，涎腺良性肿瘤与普通型多形性腺瘤间、涎腺恶性肿瘤与生长活跃型多形性腺瘤间差异无统计学意义。结论 涎腺生长活跃型多形性腺瘤与涎腺恶性肿瘤有相似的MMP-2、9和TIMP-1、2表达特征，而普通型多形性腺瘤中MMP-2、9和TIMP-1、2的表达与涎腺良性肿瘤相近。检测MMPs、TIMPs有助于判断涎腺多形性腺瘤的生物学行为。     

Intraosseous pleomorphic adenoma of the mandible: report of a case and review of the literature

Salivary gland tumors constitute approximately 3% of all head and neck tumors. The most common neoplasm involving both major and minor salivary glands is pleomorphic adenoma. Salivary gland tumors are also known to develop within jawbones probably arising from ectopic salivary tissue. Pleomorphic adenomas arising within the jaws as primary central lesions are extremely rare with only a few cases reported. Clinically and radiographically these may resemble lesions of odontogenic origin. We present a rare case of intraosseous pleomorphic adenoma of the mandible mimicking a lateral periodontal cyst along with an extensive review of literature.