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Fine-needle aspiration biopsy of adenoid cystic carcinoma of the breast: a case report.

We describe the cytologic findings of a fine-needle aspiration (FNA) breast lesion with a typical histology for adenoid cystic carcinoma. The aspirate yielded highly cellular smears with a monomorphic population of small, slightly atypical cells, arranged in multilayered groups with abundant fine intercellular metachromatic substance between cell groups. There were also fragments of fibrillar stroma in close relation to epithelial cells. The diagnosis based on the FNA material was suspicious of malignancy. The cytomorphology in this case presented a difficult differential diagnosis with pleomorphic adenoma. Features indicative of adenoid cystic carcinoma were nuclear cell hyperchromasia, the presence of small nucleoli, and scant, poorly defined, or absent cytoplasms. Differential diagnosis of breast adenoid cystic carcinoma with other entities will also be discussed.

Adenoma, Pleomorphic↗

Lymph node involvement by direct extension in adenoid cystic carcinoma. Absence of classic embolic lymph node metastasis.

Thirty-four cases of adenoid cyctic carcinoma seen at the University of Virginia Hospital from 1946 to 1974 were reviewed, with special emphasis on lymph node involvement by tumor. Lymph node involvement was found in three cases of primary tumors of the submaxillary gland, and all of the affected lymph nodes were in the immediate vicinity of the primary tumor. Two lymph nodes were involved in two of the cases, and one node was involved in the third case. In all of these lymph nodes, adenoid cystic carcinoma was present in the soft tissue surrounding the node, and the tumor extended into the node. No metastatic tumors were observed in 46 lymph nodes removed incidentally at the time of local excision of the primary tumors in 10 additional cases or in 212 lymph nodes examined after unilateral radical neck dissections in six other cases. Five autopsies in this series showed no lymph node metastases. In this series of cases adenoid cystic carcinoma only invades lymph nodes in the immediate vicinity of the primary tumor. When lymph node involvement does occur, it does not result from embolic lymph node metastasis; rather, a direct invasion of the lymph node from tumor in the perinodal soft tissue occurs. Obviously, this small study does not completely exclude the possibility of embolic metastasis; however, if it does occur, it must be extremely rare.

Carcinoma, Adenoid Cystic↗

Primary adenoid cystic carcinoma of the esophagus: report of a case.

A case of primary adenoid cystic carcinoma of the esophagus is reported. A 51-year-old male patient had a tumor in the lower third of the esophagus which was incidentally found during an examination for cholelithiasis, and resected successfully. The tumor exhibited a polypoid appearance covered by normal esophageal epithelium, localized entirely in the submucosal layer, and morphologically identical to adenoid cystic carcinoma in the salivary glands. The patient is still alive and well three and one-half years after surgery. This seems to be a typical case of adenoid cystic carcinoma of the esophagus arising from the submucosal esophageal gland.

Aged↗

Adenoid cystic carcinoma of salivary glands. A study of 61 cases with clinicopathologic correlation.

Sixty-one cases of adenoid cystic carcinoma of the head and neck region, excluding the ear canal, lacrimal glands, larynx, esophagus, and trachea, were studied, and their different clinicopathologic aspects were analyzed. Adenoid cystic carcinoma occurred more commonly in the minor salivary glands; the palate was affected in 31% of the cases. The fifth decade of life was the age at which patients were most commonly affected, and there was a slight predominance of white women. In most patients a mass was the main complaint; in 63% the duration of symptoms was 1 year or less. Forty-one patients had Stages 3 or 4 disease when first seen, and 51.7% of the patients died of disease, with a mean survival period of 35.4 months. Three basic patterns of growth, solid, cribriform, and tubular, were identified in the histopathologic examination of the cases. Other pathologic aspects analyzed were cellular pleomorphism, mitotic activity, necrosis, vascular invasion, and perineural infiltration. The study revealed a positive correlation between location of the tumor, clinical staging, duration of symptoms, and histologic pattern of growth with the prognosis of the lesion. Tumors located in the minor salivary glands, those in which the duration of symptoms was less than 1 year, and those that showed advanced clinical staging and a predominantly solid pattern of growth had an extremely poor prognosis. Surgery is the treatment of choice of adenoid cystic carcinoma, and microscopically free surgical lines of resection must be obtained. Radiation therapy, although not curative, plays an important role in prolonging survival.

Adolescent↗

Histogenesis of adenoid cystic carcinoma of the salivary glands. Light and electronmicroscopic study.

This ultrastructural study, based on 12 cases of adenoid cystic carcinoma of the major and minor salivary glands, was conducted to determine the role and extent of participation of myoepithelial cells in their histogenesis. The tumors were composed of four major cell types; intercalated duct, myoepithelial, secretory, and pluripotential reserve/stem cells. The cellular composition of adenoid cystic carcinoma is similar to that in the "terminal tubule" complex stage of a developing salivary gland except that in the tumor the pluripotential reserve/stem cells differentiate predominantly along the intercalated duct cell line rather than secretory cells as in the acinic cell carcinoma. Furthermore, adenoid cystic carcinoma appears to contain a far greater number of myoepithelial cells than acinic cell carcinomas.

Adult↗

Adenoid cystic carcinoma of the breast: diagnosis by fine-needle aspiration.

Adenoid cystic carcinoma rarely occurs as a primary breast malignancy. When seen in fine-needle aspiration (FNA) material, it is identical to its counterpart in the salivary glands. We report six cases diagnosed by aspiration in patients aged 32-82 yr. The smears were highly cellular and featured extracellular spheres of metachromatic material. These were surrounded by small, uniform cells with bland nuclei and very little cytoplasm. Numerous similar cells occurred singly in the background. The cytologic differential diagnosis of adenoid cystic carcinoma includes other entities that produce extracellular metachromatic spheres including collagenous spherulosis. We describe and illustrate similar material originating in inspissated secretions or stromal fragments from fibrocystic change and fibroadenoma, as well as infiltrating ductal carcinoma. Diagnostic criteria for adenoid cystic carcinoma and the clinical relevance of distinguishing this rare tumor from typical infiltrating ductal carcinoma are discussed.

Adult↗

DNA content in adenoid cystic carcinomas.

BACKGROUND: A retrospective study was performed on 51 patients with adenoid cystic carcinomas to see whether DNA ploidy, tumor stage, and histopathologic grading correlated with prognosis. METHODS: Histopathologic grading was performed according to Szanto et al and DNA content was estimated from archived material using the technique by Hedley et al. RESULTS: Thirty-nine tumors were DNA diploid and 12 were DNA aneuploid. Histologic grade III was more often associated with DNA aneuploidy than the lower grades (p = 0.011). DNA ploidy also correlated with clinical stage (p = 0.011). Log-rank analysis and Cox regression analysis of treatment failures revealed significant findings for S-phase value and DNA ploidy. CONCLUSIONS: Our results indicate that DNA ploidy estimations, S-phase value, and histologic grading are prognostic factors in adenoid cystic carcinomas. These examinations should therefore be incorporated in the evaluation of patients with adenoid cystic carcinomas.

Adult↗

Adenoid cystic carcinoma of Bartholin's gland: a review of the literature and report of a patient.

We report the case of a 77-year-old white female with adenoid cystic carcinoma of Bartholin's gland. To date only 45 cases of adenoid cystic carcinoma of Bartholin's gland have been reported in the world literature. The longest reported survival is 27 years. This patient was originally diagnosed 33 years ago and presented with her fourth recurrence. She was treated with radical surgery and did well for 6 months, but later died secondary to renal failure. Adenoid cystic carcinoma of the Bartholin's gland is a rare tumor of the vulva. When diagnosed the treatment should be tailored to the patient. When margins are found to be positive, adjuvant radiotherapy may prove to be beneficial.

Aged↗

On the isolation of mast cells from human adenoids and tonsils.

Human adenoids and tonsils were disintegrated mechanically and the cells dispersed by passage through a stainless-steel screen in EDTA-containing buffer. Collagenase digestion did not increase the yield of adenoidal cells. The mast cell content of the cell suspensions was in the range of 1-10 mast cells/10(4) cells with an estimated mean of 1-2 mast cells/10(4) cells, a value considerably below previous reports on adenoidal cell suspensions. The mast cell content was determined by staining with toluidine blue at low pH (to prevent interference by phagocytes). The mast cell count as assessed by alcian blue staining and by fluorescence microscopy after FITC-anti-human IgE binding was similar. Various attempts to enrich the cell suspension (i.e. by differential centrifugation, by gradient centrifugation on Ficoll or Ficoll-Hypaque and by velocity sedimentation at unit gravity) all gave negative results.

Adenoids↗

[Adenoid cystic sweat gland carcinoma. A clinicopathologic and immunohistochemical study].

Adenoid cystic carcinoma of the skin is one of the rarest sweat gland tumours. We have studied two cases of adenoid cystic sweat gland carcinoma clinically, histologically and immunohistochemically. One tumour was located on the occiput of an 18-year-old male patient, while the other developed on the back of a 49-year-old woman. Histologically, both carcinomas were characterized by basaloid tumour cells showing the typical adenoid-cystic growth pattern. The cells were arranged in solid, cribriform, tubular and cystic aggregates, which were surrounded by mucoid, Alcian blue material. Immunohistochemically, the tumour cells showed coexpression of cytokeratins typical of stratified epithelia (CK1/5/10/14) and cytokeratins of the simple epithelial type, namely CK7, CK8, CK18, and CK19. Coexpression of these cytokeratins is usually observed in normal fetal sweat glands, but not in adult sweat glands.

Adolescent↗

[Rare site of an adenoid cystic carcinoma in the nasopharynx].

We report the case of a 63-year-old patient suffering from a nasopharyngeal adenoid cystic carcinoma. She presented with increased oral secretion and pharyngeal irritation, Horner's syndrome, and trigeminal neuralgia. Magnetic resonance imaging scans revealed a tumor of the nasopharyngeal space invading the right cranial base. Lymph node metastases were clinically excluded. The histological sample confirmed an adenoid cystic carcinoma, which was therapeutically treated with adequate radiotherapy. Based on the presented case report and a review of the literature, we discuss the diagnosis and treatment of adenoid cystic carcinomas of the nasopharynx.

Biopsy↗

A potential role for arginase in recurrent infections and hypertrophy of tonsillar and adenoidal tissue.

Since arginase has been found to be an arginine-depleting and nitric oxide synthase-regulating enzyme, the present study was devised to examine hypertrophied and infected tonsil and adenoid arginase activity in relation to a metabolic arginase-nitric oxide pathway and its association with disease processes. Tissues were taken from 32 children undergoing adenotonsillectomy. There was a statistically significant difference between the two tissue enzyme activities, with tonsillar arginase activity being higher than the corresponding adenoidal tissue (P < 0.005). This suggests a potential role for tissue arginase activity as an outcome module and a contributing factor in chronic recurrent infection and hypertrophy of tonsillar and adenoidal tissues.

Adenoids↗

The effect of laminin and its peptide SIKVAV on a human salivary gland adenoid cystic carcinoma cell line.

We have previously demonstrated that laminin modulates the expression of adhesion molecules in an adenoid cystic carcinoma cell line (CAC2 cells). We are currently studying whether laminin can induce modifications in the overall morphology of CAC2 cells. These cells were grown in a three-dimensional preparation of laminin-1. Phenotype differences were assessed by light and transmission electron microscopy. CAC2 cells grown inside laminin-1 formed ductlike and pseudocystic structures. Based on our findings we suggest that laminin is a key regulator of tubular and pseudocystic patterns of adenoid cystic carcinoma. We also analyzed the effect of a molecular domain of laminin-1, the peptide SIKVAV (Ser-Ile-Lys-Val-Ala-Val) on CAC2 cells. This peptide was chosen because it is effective in cell proliferation and differentiation, and because it has never been tested before in salivary gland neoplasms. When CAC2 cells were grown inside SIKVAV-enriched laminin-1, only pseudocystic structures were observed. Since no ductlike structures were observed in samples treated with SIKVAV, we may assume that this peptide is at least one of the molecular domains of laminin responsible for the pseudocystic pattern observed in adenoid cystic carcinoma. Function disturbing experiments strongly suggested that the integrin alpha3beta1 play a role in the effect of laminin on CAC2 cells.

Antibodies, Blocking↗

Perlecan (heparan sulfate proteoglycan) gene expression reflected in the characteristic histological architecture of salivary adenoid cystic carcinoma.

In order to determine the role of the basement membrane-type heparan sulfate proteoglycan (HSPG), known as perlecan, in the formation of the characteristic cribriform 'structures of salivary adenoid cystic carcinomas, the mode of expression of mRNA for the core protein of HSPG was investigated by using in situ hybridization (ISH) both in surgical specimens and in a cell system (ACC3) of adenoid cystic carcinomas. In the surgical specimens, the mRNA for the HSPG core was more intensely expressed in solid tumor cell nests, especially in smaller ones. Within the nests, the signals were detected almost exclusively in cuboidal cells forming small pseudocysts. In contrast, signals were absent in flat cells forming large pseudocysts or in carcinoma cell nests attaching to the peripheral nerves or blood vessels. In normal salivary gland tissues, myoepithelial cells expressed the mRNA at a high level, but acinar and duct epithelial cells did not. In the time-course experiment of ACC3 cells, signals for HSPG core increased with time and reached the maximum on day 4, decreasing thereafter in a culture condition in which cells reached confluence in a week. The results indicate that HSPG is biosynthesized by adenoid cystic carcinoma cells which are in the proliferation phase, and that tumor cells producing HSPG tend to form initial structures of stromal pseudocysts.

Carcinoma, Adenoid Cystic↗

Long-term local control of recurrent adenoid cystic carcinoma in the parotid gland with radiotherapy and intraarterial infusion chemotherapy.

We report a case of postoperative recurrence of adenoid cystic carcinoma in the left parotid gland treated with radiotherapy and arterial infusion chemotherapy. A 52-year-old woman had a history of surgical resection for an adenoid cystic carcinoma arising from the left parotid gland 25 years before. Despite two reoperations for local recurrence after the initial surgery, she had a third local recurrence in the remnant of the left parotid gland. The patient was treated with a concurrent combination of radiotherapy (60 Gy/30F) and intraarterial infusion chemotherapy with carboplatin (750 mg/30 days) from which she obtained a complete response. The patient has remained free of local progression 54 months after treatment. Radiotherapy and arterial infusion chemotherapy is effective treatment for local recurrence of adenoid cystic carcinoma in the head and neck.

Antineoplastic Agents↗

Adenoid cystic carcinoma of the salivary glands: an immunohistochemical analysis.

Adenoid cystic carcinoma is a specific variant of adenocarcinoma with a characteristic cribriform appearance. The tumor may arise from salivary glands and various other sites, but the origin and cellular composition of this unique neoplasm have been controversial. A potential use of immunohistochemistry is to provide additional information on the origin of various cellular components of tumors by comparing them with corresponding normal tissues. Immunohistochemical distributions of carcinoembryonic antigen (CEA), epithelial membrane antigen (EMA), keratin, low molecular weight keratin (54 kd), S100 protein, muscle-specific actin, laminin, and type IV collagen were evaluated in 20 adenoid cystic carcinomas arising in major and minor salivary glands. Anti-CEA, anti-EMA, anti-keratin, and anti-S100 antibodies strongly stained cells lining true lumina. Muscle-specific actin, a marker for myoepithelial cells, was found in lining cells of pseudocysts, in tumor cells proper, and in nonluminal cells with a tubular growth pattern. A monoclonal antibody against 54 kd keratin stained almost all cells in the neoplasms. In pseudocysts, replicated basal lamina reacted with antisera to laminin and to type IV collagen. The present study demonstrates that there are at least two populations of tumor cells in adenoid cystic carcinoma: luminal cells that express CEA and EMA, thus indicating their ductal character, and nonluminal cells that express muscle-specific actin characteristic of myoepithelium.

Adult↗

The functional role of the adenoids in speech.

Illustrative cases are presented showing a variety of interrelationships between the adenoids and the activity of the velopharyngeal valve in speech. The cases presented were selected from a group of 1000 patients referred because of suspected velopharyngeal diseases. When appropriate, complete velopharyngeal assessment was made including otolaryngological speech and hearing examination, polysomnography, nasendoscopy, multiview videofluoroscopy and cephalometry. New observations are described which further elucidate the mechanism by which the adenoids may change the mechanism of velopharyngeal valving and consequently speech patterns. In conclusion, procedures involving the adenoids and tonsils and surgical correction of velopharyngeal valve abnormalities to improve respiratory function must be performed in a manner which ensures preservation of normal speech activity. Similarly, surgical correction of velopharyngeal valve abnormalities to improve speech activity must preserve its respiratory function. The velopharyngeal valve and the adeno-tonsils must be considered together whenever diagnosis and a therapeutic intervention of either of them is considered. A clinical method for patient evaluation, patient management and the development of a rational therapeutic approach is presented.

Adenoids↗

Radiation therapy in adenoid-cystic carcinoma.

Between 1949 and 1977, 74 patients with adenoid-cystic carcinoma of various head and neck sites were treated by radiation therapy at Memorial Sloan-Kettering Cancer Center. Radiation therapy alone was employed in 49 patients for recurrent, unresectable disease, and in 25 patients it was given as an adjunct to surgical resection. Among the 49 patients treated with radiation therapy alone, tumor regression was seen in 47 (96%). However, 44 of the 47 (93.5%) subsequently relapsed locally. Relapse occurred within 18 months in one-half of the patients and within 5 years in all of them. Of the 25 patients who received adjunctive radiation therapy about one-half relapsed locally within five years. There were 9 patients in this group, however, whose field size exceeded 8 X 8 cm and the dose of radiation also exceeded 4500 rad: 88% of these patients remained relapse-free at 5 years, compared with only 22% of the other 16 whose dose, or field size, or both, were inadequate by comparison. These data suggest that when irradiation is employed for advanced, inoperable adenoid-cystic carcinoma, it offers useful palliation but is rarely, if ever, curative. Postoperative irradiation, on the other hand, might improve the local control and the survival in patients with operable adenoid-cystic carcinoma who are at high risk for relapse, but only if the field size and the dose are adequate.

Adult↗