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[Adenoid cystic carcinoma of the trachea and bronchi. Presentation of 2 cases and review of the literature].

Adenoid cystic carcinoma is a rare but insidious tumor of the trachea and bronchi. It is infiltrative and tends to recur at the same site. Diagnosis is often late, generally when a large part of the bronchial opening is occluded and with extensive invasion of submucosal tissue, making elective surgery difficult and complementary radiotherapy necessary in most cases. The prognosis, however, is fairly good, with survival after 5 years in 75% of patients. We present cases diagnosed recently at our hospital, one with a large longitudinal tracheal tumor and the other with a located in the carina. The literature is reviewed.

Adult↗

Adenoid cystic carcinoma of the major and minor salivary glands. A clinicopathological study of 17 cases.

Local recurrence and distant metastasis were studied in relation to treatment modality and histological findings in 17 patients with adenoid cystic carcinoma of the major and minor salivary glands. Fifteen patients underwent surgical resection with or without postoperative irradiation and the others were treated by irradiation alone. Local recurrence developed in nine patients (52.9%), mostly within two years of the treatment. Incomplete removal was the major reason for the failure to control the primary lesion. Postoperative irradiation was of value in preventing recurrence in patients who demonstrated microscopic residual disease at the surgical margins, whereas it was not effective if gross residual tumour was recognized. Direct surgical intervention on the tumour tissue could have been the possible cause for the distant metastasis that developed in 10 (58.8%) of the 17 patients. Surgical excision with a wider safety margin than the visibly affected area, followed by postoperative irradiation and chemotherapy was essential to obtain a better prognosis.

Adult↗

Extracellular matrices expression in invasion area of adenoid cystic carcinoma of salivary glands.

Adenoid cystic carcinoma (ACC) is a salivary malignant tumor with poor long-term prognosis, that is known to have predilection for invasion of the adjacent stroma and neural tissues. This carcinoma has shown a high incidence of recurrence and distal metastasis. Invasive carcinomas have been associated with the distributions of extracellular matrices (ECM). Cell proliferation as a marker of tumor growth has been related to poor prognosis in oral carcinomas. Immunohistochemical analysis of 15 cases of ACC was done using antibodies to laminin, type IV collagen, fibronectin, tenascin and anti-proliferating nuclear antigen (PCNA). Laminin and type IV collagen were totally or partially absent in the ACC invasive areas. Tenascin was expressed in the stroma and cytoplasm and was associated with tumor cell proliferation. It can be concluded that basement membrane represents a barrier that is lost during cell invasion and tenascin may be involved in the detachment of cancer cells, increasing the invasive potential of ACC.

Adult↗

An immunohistochemical study of the distribution of lysozyme, lactoferrin, alpha 1-antitrypsin and alpha 1-antichymotrypsin in salivary adenoid cystic carcinoma.

The immunohistochemical expression of lysozyme (Ly), lactoferrin (La), alpha 1-antitrypsin (alpha 1-AT), and alpha 1-antichymotrypsin (alpha 1-Ach) was described, and their distributions were compared to each other in 28 cases of adenoid cystic carcinoma (ACC) of the salivary glands. ACC materials were obtained from the parotid gland (7), the submandibular gland (4), the sublingual gland (8), and minor oral salivary glands (9). Histopathologically, ACC was classified into cribriform (14), tubular (3), and basaloid or solid patterns (11). Positive staining for Ly was found in 1 case of solid ACC in the sublingual gland; La was found in 4 cases (2 cribriform, 1 tubular, 1 basaloid) in the sublinguals (3) and parotid glands (1); alpha 1-AT was found in 6 cases and alpha 1-Ach in 17 cases. The immunohistochemical localization of Ly and La was usually confined to luminal tumor cells of tubulo-ductal structures, irrespective of the pathologic types. Positive staining for alpha 1-AT and alpha 1-Ach appeared in tumor cells of cribriform, tubular and solid ACC. Tumor cells with positive La staining coincided with a positive reaction to alpha 1-AT and alpha 1-Ach, and tumor cells with alpha 1-AT positive deposition were also positive for alpha 1-Ach. The contents of pseudocysts in the cribriform pattern showed a positive reaction to La, alpha 1-AT, and alpha 1-Ach. Of the 28 cases of ACC, positive expressions for Ly, La, alpha 1-AT and alpha 1-Ach were found with a high frequency of alpha 1-Ach staining (17 in 28 cases were positive). In sublingual ACC (8), 7 cases were positive for immunohistochemical reactions. Co-expression or simultaneous expression for Ly, La, alpha 1-AT, and alpha 1-Ach in ACC suggest that tumor cells are protected from proteolysis or degradation.

Carcinoma, Adenoid Cystic↗

Assessment of proliferative activity using the MIB1 antibody help to distinguish polymorphous low grade adenocarcinoma from adenoid cystic carcinoma of salivary glands.

Salivary polymorphous low grade adenocarcinoma (PLGA) and adenoid cystic carcinoma (AdCC) bear a superficial histological and immunophenotypic resemblance to each other, but can usually be separated by conventional microscopic examination. However, this is not always so, such as with PLGAs displaying plentiful cribriform structures, and when only limited tissue from small biopsies is available for study. Twenty-one cases of PLGA and 20 cases of AdCC from the surgical pathology files in Plzen, Exeter and Helsinki were incubated with the MIB1 antibody using the supersensitive avidin-biotin peroxidase technique after microwave pretreatment. The antibody recognizes the cell cycle associated antigen Ki-67 on routinely processed formalin-fixed paraffin-embedded tissues. The percentage of positively stained tumor cell nuclei constituted the MIB1 index. It was found that the PLGAs had a mean MIB1 index of 2.4% (range 0.2-6.4), while the AdCCs had a mean value of 21.4% (range 11.3-56.7). Within the PLGA group, the 12 tumors which did not form micropapillae had a somewhat lower mean MIB1 index (1.4%) than those nine in which such structures constituted a significant but minor component (3.4%). These results indicate that the MIB1 index was significantly higher in the AdCCs than the PLGAs and that the figures showed no overlap zone. We conclude, therefore, that immunohistochemical staining with the MIB1 antibody appears to be a potentially useful supplementary diagnostic tool in differentiating difficult cases of PLGA from AdCC.

Adenocarcinoma↗

Feasibility and toxicity of combined photon and carbon ion radiotherapy for locally advanced adenoid cystic carcinomas.

PURPOSE: To investigate clinical feasibility and toxicity of combined photon and carbon ion radiotherapy in locally advanced adenoid cystic carcinomas (ACC) within a prospective Phase I/II trial. METHODS AND MATERIALS: Between September 1998 and April 2002, 16 patients with histopathologically proven ACC and residual macroscopic tumor were treated with combined photon RT and a carbon ion boost to the macroscopic tumor. Median total tumor dose within the gross tumor volume (GTV) was 72 GyE. Photon radiation therapy (RT) consisted of fractionated stereotactic RT in 7 patients; 9 patients received stereotactic intensity-modulated RT. Carbon ion boost was delivered by intensity-controlled raster scanning at the heavy ion synchrotron (SIS) at the Heavy Ion Research Center (GSI) in Darmstadt. RESULTS: Median follow-up was 12 months. Three patients developed locoregional recurrences 9, 11, and 24 months after RT, respectively. Actuarial local control rates were 80.8% and 64.6% at 1 and 3 years, respectively. Overall survival rates were 100% and 83.3% at 1 and 3 years, respectively. Acute side effects greater than Common Toxicity Criteria (CTC) Grade 2 were observed in 2 patients; no patient developed late effects > CTC Grade 2. CONCLUSIONS: Combined photon and carbon ion RT is feasible and effective in patients with locally advanced ACC. Acute and late toxicity is moderate with respect to the delivered tumor doses and in accordance with the radiobiologic modeling. A Phase III trial is designed.

Adult↗

Clinical study of adenoid cystic carcinoma of the head and neck.

OBJECTIVE: We examined prognostic factors and outcome of the primary treatment in patients with adenoid cystic carcinoma (ACC) of the head and neck. METHODS: Twenty patients with ACC of the head and neck who had been treated in our institution from 1985 to 1998 were enrolled in this study. Disease-specific survival rate was analyzed by the Kaplan-Mejer method, and the log-rank test was applied to compare the survival rates. RESULTS: The overall 5- and 10-year survival rates determined by Kaplan-Meier analysis were 81 and 57%, respectively. Patients with major salivary gland ACC obtained the best 10-year survival rate (83%), while those with paranasal sinus ACC had the worst survival rate (33%). Predominance of the solid component on pathological examination might indicate a worse prognosis. Our study revealed that postoperative radiotherapy could yield better control of the lesion focus. Chemotherapy failed in some patients and was not dramatically effective by itself. CONCLUSIONS: The long-term prognosis of ACC was poor. Long-term follow-up is necessary for better prognosis of patients treated with radical treatment regimens.

Adult↗

Adenoid cystic carcinoma of the lacrimal gland with wide and severe myoepithelial differentiation.

Adenoid cystic carcinoma (ACC) of the lacrimal gland is the second most common epithelial tumor for which different biologic courses can be predicted by histologic criteria. Three main types of growth patterns, cribriform; tubular; and solid have been identified. Tumors with solid components frequently follow a more aggressive clinical course and show worse prognosis than those with other patterns. We herein report a case of ACC with wide and severe myoepithelial differentiation arising from the lacrimal gland and presenting with aggressive clinical behavior. Postoperative radiotherapy may be the treatment of choice to control residual lesions and provide long-term survival even in the case of incomplete resection. Despite extensive surgery and radiation therapy, the prognosis of these tumors, especially with solid components, remains extremely poor. Accurate diagnosis is important because tumor histopathology is generally believed to be the most significant factor in patient survival.

Carcinoma, Adenoid Cystic↗

Unusual case of myoepithelioma associated with adenoid cystic carcinoma of the parotid gland.

Tumors of the salivary glands comprise less than 3% of all neoplasms of the head and neck, with synchronous parotid tumors of differing histologic types being extremely rare. We present a 71-year-old woman with a myoepithelioma associated with an adenoid cystic carcinoma of the left parotid gland. The 48 previously reported cases of synchronous occurring parotid gland tumors of differing histologic types are reviewed.

Aged↗

Long-term results of surgery for adenoid cystic carcinoma of the trachea and bronchi.

AIMS: Over a period of 26 years, 16 patients (9 women, 7 men) underwent surgery for primary adenoid cystic carcinoma (ACC) of the trachea and bronchi. The median age at diagnosis was 41.4 years (range 25-67). Nine tumours were located in the bifurcational area, five in the trachea, one in the middle-lobe bronchus and one in the parenchyma of the left lower lobe. METHODS: Surgical procedures were as follows: three tracheal transversal resections; five resections of the distal trachea including the bifurcational region, followed by bifurcational reconstruction; two right-sleeve pneumonectomies; three left-sleeve pneumonectomies; two lobectomies; and one explorative sternotomy. RESULTS: Eleven patients were available for follow-up at least 5 years after surgery. Three of these patients (27%) had local recurrence 155+/-30 (range 120-175) months after surgery. Distant metastases occurred in six patients (55%) after a median time interval of 96+/-68 (range 24-180) months after surgery. CONCLUSIONS: Five-year and 10-year survivals were excellent, 79 and 57% respectively, but the long-term outcome was poor due to late local recurrences and late metastatic spread. It is not yet certain whether a cure can really be achieved in ACC.

Adult↗

Selection of adenoid cystic carcinoma cell clone highly metastatic to the lung: an experimental study.

Lung metastasis is very common in patients with adenoid cystic carcinoma (ACC). The purpose of the present study is to establish a model for an experimental study of ACC metastatic mechanisms and antimetastatic procedures. After five repeated selection in vivo, combined with an in vitro cloning technique and analysis of platelet aggregation activity, a clone (Acc-M) highly metastatic to the lung was selected from an ACC cell line (Acc-2). The metastatic rate was 96% vs 18% for Acc-M and the Acc-2 cell line. The metastatic rate and the weight of the lung with metastases correlated positively with platelet aggregation activity. The present study might be important for research into mechanisms of ACC metastases.

Animals↗

The significance of perineural spread in adenoid cystic carcinoma of the major and minor salivary glands.

Patient survival, local recurrence and distant metastasis were studied in relation to the pathological finding of perineural spread in 37 patients with adenoid cystic carcinoma of the major and minor salivary glands. All patients underwent a combined surgical and radiotherapeutical treatment. The overall incidence of perineural invasion in primary resection specimens was 52.6%. The 5-year actuarial survival rate for patients with perineural invasion was significantly lower (p less than 0.001) than for those without (36.9% versus 93.8%). In 26 patients with resection margins free of tumour, recurrences developed in 9/11 (81.8%) of the patients with perineural invasion as opposed to 4/15 (26.7%) of the patients without perineural invasion (p = 0.005). In the same group with resection margins free of tumour, distant metastasis developed after the primary treatment in 4/10 (40.0%) of the patients with perineural invasion, while none of the 14 patients without perineural invasion experienced distant metastasis (p less than 0.0002). The incidence of perineural invasion increased with a higher stage of the primary tumour.

Adult↗

Factors influencing survival rate in adenoid cystic carcinoma of the salivary glands.

Ninety-one cases of adenoid cystic carcinoma (ACC) of the salivary glands with more than ten years' follow up were studied to investigate factors influencing the survival rate of patients, which vary according to site, histological type, clinical stage and nature of therapy. The data were statistically analysed for survival curves. Log rank tests were employed to assess the statistical significance of various groups. As a result, it may be concluded that tumour site, clinical stage and histological type are the important factors influencing the prognosis. ACC of the palate and parotid, early clinical stage, glandular/tubular histological type, and tumour without nerve involvement had the best prognosis. ACC in the submandibular gland, maxillary antrum and tongue, advanced clinical stage (stage III and IV), solid histological type, and tumour with nerve involvement had a poor prognosis.

Adult↗

Adenoid cystic carcinoma of the infraspheno-temporal fossa. Latero-facial resection combined with multiple osteotomies.

Two cases of Adenoid Cystic Carcinoma (ACC) of the infrasphenotemporal fossa with invasion of the floor of the middle fossa are presented. Preoperative studies included CT-scan and cervical and cerebral angiography. The exposure and operative management comprised a latero-facial approach combined with multiple exposure osteotomies of the ascending mandibular ramus, orbitozygomatic region and a frontosphenotemporal craniotomy. This approach provides wide exposure of the posterolateral orbit, sphenoid wing and infratemporal and pterygomaxillary fossa. One of our patients died from massive recurrence in proximity to the cavernous sinus and the other is free of disease after 30 months.

Carcinoma, Adenoid Cystic↗

The adenoid cystic carcinoma. Special aspects of its growth and therapy.

In spite of its rare incidence the adenoid cystic carcinoma must be treated with respect because of its hidden malignant character. In 5 out of 21 patients recurrences occurred much longer than 5 years after the first operation. The extension of this tumour along branches of the trigeminal nerve could be well demonstrated in 7 of our patients where we were able to produce histological proof or radiographic evidence. The theory of tumour growth along the "tissue of least resistance" is illustrated by a special case report. As for the therapy, ultra-radical surgery has proved to be the most effective so far, whereas radio- or chemotherapy were only useful as palliative measures.

Adult↗

Adenoid cystic carcinoma of the trachea and bronchus--a clinicopathologic study with DNA flow cytometric analysis and oncogene expression.

OBJECTIVE: Adenoid cystic carcinoma (ACC) of the tracheobronchial tree is quite uncommon. The clinicopathologic analysis and the therapeutic outcomes of tracheobronchial ACC have been reported earlier. However, their biological behavior should differ from other tracheal neoplasms. Thus, DNA flow cytometric analysis and biomarkers of p53, HER-2/neu and COX-2 for tracheobronchial ACC were investigated in order to evaluate their clinicopathological significance. METHODS: Between 1985 and 1999, nine patients with tracheobronchial ACC were included for the study. All the patients had pathologically confirmed ACC. Five were male and four were female. Eight patients underwent surgical resections. Seven paraffin embedded tumors from six patients were available for DNA flow cytomeric analysis and immunohistochemical staining of p53, HER-2/neu and COX-2. RESULTS: Histologically, nine pathologic specimens from eight surgical patients (including one patient received operation twice) showed one grade I, five grade II and three grade III. The mitotic activity, lymphatic invasion and vascular invasion were more frequent in advanced grading tumor. The higher grade tumors seemed to be associated with a higher synthetic phase fraction (SPF). Immunohistochemically, except for one grade II tumor showing positive expression of HER-2/neu, all the seven tumorous samples revealed negative expressions of p53, COX-2 and HER-2/neu. The patient with positive HER-2/neu tumor had distant metastases 4 years after surgery. CONCLUSIONS: Complete surgical resection may provide best survival for tracheobronchial ACC. The DNA ploidy and SPF may correlate with tumor grading or metastasis. The overexpressions of HER-2/neu, p53 and COX-2 may impact the prognosis in patients with stage I non-small cell lung cancer, but did not express difference in our patients.

Adult↗

Tumor-doubling time and onset of pulmonary metastasis from adenoid cystic carcinoma of the salivary gland.

Adenoid cystic carcinoma (ACC), an uncommon malignancy in the head and neck region, invades diffusely and often metastasizes to the lung, although the growth rate is very slow. A retrospective study was conducted in 30 patients with ACC to ascertain the frequency of pulmonary metastasis, the doubling time of metastatic tumor deposits, and the time of onset for pulmonary metastasis. The following results were obtained: (1) Of 30 patients with ACC, 21 had pulmonary metastases (4 initially and 17 during observation), 7 were free of metastases but have not been observed for 5 years, and 2 were free of metastases for more than 5 years but less than 10 years after the initial treatment. The cumulative metastasis rate at 5 and 10 years for this group of patients was 70% and 100%, respectively. (2) Patients with T1 or T2 tumors that have a tubular or cribriform histopathologic pattern showed pulmonary metastases about 20 months later than those with T3 or T4 tumors and a solid pattern. However, the final metastasis rate did not differ between the 2 groups after a long period. (3) The tumor doubling time of the metastatic deposits of ACC was 86 to 1064 days with an average of 393 days, which was much longer than that of most other malignant neoplasms reported previously. (4) The time of onset of pulmonary metastasis was calculated to be much earlier (average of 227 months) before the first visit. These findings suggest that the treatment method for ACC should be chosen with the consideration that many of the patients may have occult pulmonary metastases at the time of their initial evaluation.

Carcinoma, Adenoid Cystic↗