Response to paclitaxel in adenoid cystic carcinoma of the salivary glands

BACKGROUND: Paclitaxel is not considered to be an active drug in adenoid cystic carcinoma (ACC) of the salivary glands. We report 2 consecutive cases of patients with ACC who responded to paclitaxel. METHODS: The patients were: (1) a 58-year-old man with recurrent ACC who developed pulmonary metastases, had progressive disease after a good response to first-line chemotherapy, and then achieved a partial response to weekly single- agent paclitaxel; and (2) a 46-year-old woman with extensive thoracic ACC metastases who achieved a significant response after 2 cycles of paclitaxel chemotherapy. RESULTS: The first patient died of progressive disease approximately 4 months after completing paclitaxel therapy, and the second patient had disease control after 6 cycles of paclitaxel. CONCLUSIONS: Systemic weekly paclitaxel produced a significant response in 2 patients with ACC of the head and neck, and its use in this disease merits further study.     

Imatinib mesylate as treatment for adenoid cystic carcinoma of the salivary glands: report of two successfully treated cases

BACKGROUND: Adenoid cystic carcinoma (ACC) is a malignant neoplasia of the salivary glands that is treated primarily by surgery. Local control and survival are usually compromised despite surgery. Expression of KIT tyrosine kinase is involved in the pathogenesis of ACC. Imatinib mesylate is a potent inhibitor of KIT tyrosine kinase, so we explored the possibility that ACC could be a potential target for this drug. METHODS: We report two cases of unresectable ACC treated with imatinib mesylate in the context of recurrent disease (case 1) and locally advanced tumor at its initial presentation (case 2). RESULTS: Both patients responded well to treatment with imatinib mesylate. Significant regression of recurrent disease (case 1) resulted in a successful salvage surgical resection; the locally advanced tumor (case 2) had an excellent response to treatment, but, unfortunately, the patient refused salvage resection. CONCLUSION: This is the first time ACC is reported to respond to imatinib mesylate. Studies in which more patients are enrolled in controlled clinical trials are needed to confirm this observation.     

Lung metastasis resection of adenoid cystic carcinoma of salivary glands

Background: Adenoid cystic carcinoma is a rare tumour originating from the exocrine mucous glands, known for its high propensity for distant metastases. The value of lung metastasis resection from adenoid cystic carcinoma of salivary glands origin is evaluated. Methods: A retrospective study was conducted on patients undergoing surgery for primary adenoid cystic carcinoma of the salivary glands between 1982 and 2006. Patients were excluded who had primary tumour macroscopic incomplete resection or were lost at follow-up. From a database of 50 eligible patients, 27 were identified as having presented a tumour recurrence during follow-up; in 20 it was first diagnosed in the form of distant metastases, and in 7 in the form of loco-regional recurrence. Nine patients who presented isolated lung recurrence underwent complete lung metastasectomy. Demographic data, pathologic characteristics and operative and postoperative record were reviewed, as well as updated survival. Results: Twenty-six men and 24 women with a median age of 57 years (range 33–79) underwent radical surgery for adenoid cystic carcinoma during the study period. In 20 patients, at a median free interval time of 3 years (range 1–12), a distant metastasis relapse was observed. Nine patients with a median free interval time of 5 years (range 1–12) underwent lung metastasectomy: five had single metastasis resection, one multiple mono-pulmonary and three multiple and bilateral. In six of these patients a new disease recurrence was noted: four patients underwent further lung metastasectomy, but in all of them progression of the disease was observed. Mean survival of the population as a whole resulted as being 16 years (SE = 1.4) with an actuarial survival of 77% at 5 years, 66% at 10 years and 56% at 15 years. Mean survival of patients having presented with distant metastases resulted as being 11 years (SE = 2.2). Mean survival after appearance of distant metastases resulted as being 72 months (SE = 15.8) in the 9 patients treated by metastasectomy, and 62 months (SE = 15.1) in the 11 who did not have metastasis resection. Conclusions: Patients with adenoid cystic carcinoma could be frequently encountered with disease recurrence confined to the lung. The impact of complete lung metastasis resection on the course of the disease, however, is yet to be determined.     

COX-2在腺样囊性癌中的表达及意义

目的探讨COX-2在正常涎腺(Normal Salivary,NS)及涎腺腺样囊性癌(Salivary Adenoid Cystic Carcinoma,SACC)中的表达状况及其在SACC发生、发展过程中的作用及意义。方法选择32例SACC组织、11例NS组织,应用SP法免疫组织化学染色检测COX-2的表达情况,分析其在各组间表达的差异。结果 COX-2在11例NS中阳性表达2例,阳性率18.18%。在32例SACC中阳性表达25例,阳性率为78.13%。2组间差异有统计学意义(P<0.05)。在SACC患者按年龄、性别、临床分期、原发部位及病理类型等分组间比较COX-2表达差异无统计学意义(P>0.05)。在按有无侵袭比较COX-2的表达差异有统计学意义(P<0.05)。结论在SACC中,COX-2呈高表达,并与有无侵袭临床参数相关,而与患者的年龄、性别、肿瘤部位、病理类型及临床分期无明显相关性。COX-2可以作为其周围侵袭的一个指标。     

Immunohistochemical study of epidermal growth factor receptor in adenoid cystic carcinoma of salivary gland origin

BACKGROUND: Epidermal growth factor (EGF) and its receptor (EGFR) are involved in the development of salivary gland tumors. Recently, treatment modalities for EGFR inhibition have shown an enhanced clinical response in carcinomas of different locations. Adenoid cystic carcinoma (ACC) of salivary gland origin is a malignant tumor with a poor long-term outcome. If salivary gland ACC does exhibit EGFR, then immunotherapy could have a major impact on improving its prognosis. METHODS: The study consisted of 34 samples of formalin-fixed, paraffin-embedded specimens of salivary gland ACC. Specimens were stained with a mouse antihuman monoclonal antibody for immunohistochemical detection of EGFR. Overlying oral mucosa and adjacent normal salivary ducts served as internal controls. Both membrane and cytoplasmic staining were evaluated. Staining score was calculated by multiplying the percentage of positively stained tumor cells by the intensity of the staining. The highest score for a given tumor was equal to 2. RESULTS: In the final analysis, 27 of the 34 specimens were included; 7 were excluded, because the internal control did not reveal any staining. Of these 27 specimens, 23 (85%) stained positively for EGFR with a staining score of 0.05 to 1.8. Three palatal tumors attained the highest scores (one tumor, 1.2, and the remaining two, 1.8). CONCLUSIONS: Most salivary gland ACC stained positively for EGFR, and in some the staining was quite intense. On the basis of the already proven antitumoral effect of agents acting as EGFR inhibitors, it is suggested that patients with ACC might benefit from these agents, especially when surgery has failed or in those with recurrent or metastatic disease.     

Metastatic-adenoid cystic carcinoma of the major salivary glands presenting as a spinal cord tumor

An adenoid cystic carcinoma (cylindroma) of the sub-mandibular salivary gland presented 17 years after diagnosis with a compressive syndrome of the spinal cord as well as a local recurrence. This is the first reported case of such an occurence. The literature is reviewed concerning other malignant tumors of the salivary glands presenting in this way. Fifteen cases of mixed salivary-gland tumors are reviewed. Evidence of histologic change from one tumor type to another and the mechanism of spread to the vertebral column is discussed.     

The value of MYB as a prognostic marker for adenoid cystic carcinoma: Meta‐analysis

The aim of this meta‐analysis is to evaluate myeloblastosis (MYB) as a prognostic marker for patients with adenoid cystic carcinoma (ACC) with respect to MYB gene fusion, MYB protein expression, and tumor sites. We comprehensively searched PubMed, Embase, Web of Science, and Cochrane libraries. Ten studies concerning the prognostic comparisons between MYB positivity and negativity were included. The combined positive rates of MYB gene fusion and protein expression were 57.2% and 62.3%, respectively. Overall, no significant prognostic differences were observed between MYB‐positive and MYB‐negative ACCs. When further divided into MYB gene fusion and MYB protein expression subgroups, no significant differences were observed for any survival outcome (overall survival, disease‐free survival, and local control rate). Moreover, MYB also demonstrated no prognostic value in head and neck ACCs. In conclusion, the current studies reveal that MYB is not a good prognostic marker for ACC.     

Unique presentation of adenoid cystic carcinoma in postcricoid region: a case report and review of the literature

Malignant tumors of minor salivary glands are uncommon, representing only 2–4% of all head and neck malignancies. In the larynx, minor salivary gland tumors rarely occur and constitute less than 1% of laryngeal tumors. Most of the minor salivary gland tumors arise in the subglottis; however, they can also occur in the supraglottis in the false vocal cords, aryepiglottic folds, and caudal aspect of the epiglottis. The most common type of malignant minor salivary gland tumor is adenoid cystic carcinoma. In the last 20 years, only 40 cases of adenoid cystic carcinoma arising in variable locations of the larynx have been reported in the English language literature. We present a case of adenoid cystic carcinoma of the larynx arising in the inter-arytenoid and postcricoid region in a 54-year-old woman. To our knowledge, this is the first case of adenoid cystic carcinoma arising in this particular region of the larynx.     

Unexpected rapid progression of metastatic adenoid cystic carcinoma during treatment with imatinib mesylate

BACKGROUND: There is a lack of effective treatment for metastatic adenoid cystic carcinoma (ACC), a usually indolent tumor. We studied the efficacy of imatinib mesylate, a potent inhibitor of KIT tyrosine kinase, in patients with KIT-positive metastatic ACC. METHOD: Five patients with lung metastasis were treated in a pilot study with imatinib 400 mg by mouth twice a day. Mutations of c-kit and platelet-derived growth factor receptor (PDGFR)-alpha in tumors from these patients were analyzed. RESULTS: Disease progression was noted in three of five patients during the short treatment periods, ranging from 2 to 3 weeks. Three patients died of disease within 6 months. No detectable mutations were found in c-kit and PDGFR-alpha. CONCLUSION: We observed an unexpected high progression rate of metastatic ACC within short periods during imatinib treatment. Use of imatinib to treat cancers without c-kit or PDGFR-alpha mutation should be approached with caution.