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Adenoid cystic carcinoma (ACC) of the lacrimal gland is a rare tumor from the epithelial structure. It is rare in children. The authors report a case of ACC in a girl child. Extensive excision of the tumor is advised to prevent later recurrences. Local control can be better achieved by a combination of radiochemotherapy as ACC has been shown to recur event after 10 years.
A new cell line has been established from an adenoid cystic carcinoma arising in the submandibular gland of a 63-year-old woman. The cultured epithelial-like cells grew vigorously and adhered together to form a sheet. Immunohistochemical stainings for type IV collagen, laminin and fibronectin were clearly positive in the intercellular matrix and on the surface of the culture cells. Chondroitin 6-sulfate proteoglycan and heparan sulfate were also detected. Ultrastructural studies showed that the cells had abundant rough endoplasmic reticulum and a well-developed Golgi apparatus. Rough endoplasmic reticulum near the cell surface was markedly dilated, and contained material of low electron density. This cell line would be useful for biological and biochemical studies on the mechanisms by which the stromal component is formed.
Immunohistochemical studies were performed to explore the distribution of S-100 protein and its alpha, beta subunits in 76 adenoid cystic carcinomas (ACC) of the salivary glands. Histopathologically. ACC was divided into cribriform, tubular, basaloid and trabecular types which could be mixed in the same tumor. S-100 protein was usually positive in tumor cells forming cribriform structures; foci of strongly positive tumor cells were also distributed in the luminal layer of tubular structures, and in areas transitional between cribriform and tubular patterns. S-100 alpha staining was confined to some tumor cells in cribriform areas, to luminal tumor cells in tubular structures and to few tumor cells in basaloid structures. S-100 beta reaction was usually localized to luminal surfaces in a fine granular pattern in tubular and microtubular structures in a distribution somewhat similar to that in the normal salivary gland. Great heterogeneity in the immunohistochemical distribution of S-100, S-100 alpha and S-100 beta proteins was found in the various histologic types of ACC and the pattern was different from that seen in pleomorphic adenomas. It is possible that the ACC tumor cells positive for S-100 protein may be closely related to true or modified myoepithelial cells.
A case of adenoid cystic carcinoma (ACC) of the breast in a 66-year-old woman is reported herein. ACC accounts for about 0.1% of all breast cancers. Our patient presented with a small, elastic and hard mass, measuring 2.0x2.0 cm, between both outer quadrants of the right breast. Although physical examination, ultrasonography and magnetic resonance (MR) mammography suggested a benign tumor, aspiration biopsy cytology (ABC) was performed twice, and the second ABC specimen was evaluated as suspicious for breast carcinoma. Breast conserving surgery with a level II lymph node dissection was subsequently performed. There was no lymph node metastases and estrogen receptor (ER) status was negative. Light microscopy revealed various growth patterns, with the cells showing biphasic cellularity. According to immunohistochemical analyses, CEA, actin and vimentin were positive, S-100 protein was negative, and the cytokeratin reaction was partially positive. Therefore, ACC of the breast was diagnosed. Although ACC of the breast is a rare neoplasm, it should be considered in the differential diagnosis even if various diagnostic imaging studies suggest a benign tumor of the breast. Awareness of this tumor will help prevent misdiagnosis.
INTRODUCTION: Patients with adenoid cystic carcinoma (ACC) of the salivary glands in the head and neck region have been reported to benefit from neutron radiotherapy according to significant clinical experience. A prospective clinical trial on the efficacy and treatment related morbidity of fast neutron radiotherapy was performed between 1986 and 1995 at the (d + T) 14 MeV neutron generator in Münster. MATERIAL AND METHODS: 72 consecutive patients with ACC were treated with fast neutrons, 66 after surgery, 6 for primarily unresectable disease, 43/66 for macroscopic residual disease, 23/66 for unresectable recurrent disease. 45/72 tumors were localized in the minor, 27 in the major salivary glands. T-stage was in 13 pts T2, in 33 T3, in 26 T4; positive nodes were in 10 pts, M+ in 15 pts. Mean tumor volume was 89 cm3. Neutron therapy was 15.03 Gy in 3 weeks with 1.67 Gy per fraction three times per week. Individual computer assisted treatment planning was performed based on CT and/or MRI, using bolus material if necessary. Target volume was the macroscopic tumor volume with a generous safety margin. RESULTS: Complete response was achieved in 28 pts, partial response in 35 pts. Local control was observed in 73.4% after a mean observation period of 36 months. Overall and recurrence free survival was 85%/81% at two years, and 58%/53% at 5 years (Kaplan-Meier). In univariate analysis tumor volume (> 100 cm3), distant metastases, histologic subtype (solid) and neutron dose (< 15 Gy) turned out to be significant parameters for predicting outcome, in multivariate analysis tumor volume and histologic subtype remained the only significant parameters. Acute morbidity was grade III/IV (EORTC/RTOG) in 6% for skin (desquamation), in 4% for mucosa (ulceration), late morbidity (grade III/IV) in one patient with local temporal brain necrosis. CONCLUSION: According to this experience and taking into account the so far collected experience, fast neutron radio-therapy remains the treatment of choice for large and unresectable primary and recurrent ACC, and residual disease after surgery.
BACKGROUND: Tumor doses > 70 Gy are needed for local control in adenoid cystic carcinomas. These tumor doses cannot be delivered if the tolerance doses to neighboring organs at risk (OAR) are respected. This treatment planning study investigates the physical advantage of combined photon intensity-modulated radiation therapy (IMRT) plus carbon ion boost compared to photon IMRT alone. PATIENTS AND METHODS: For nine patients, treatment plans were generated using a) photon IMRT alone (integrated boost concept), and b) sum plans consisting of a photon IMRT plan and a carbon ion boost plan. 54 Gy were prescribed to the planning target volume 1 (PTV1), the boost volume (PTV2) received 72 Gy. The tolerance doses of the delineated OAR were strictly adhered to. Plan quality of IMRT plans and sum plans was compared using adequate physical parameters. RESULTS: Both therapy techniques lead to highly conformal dose distributions that allow the prescription of the desired target doses. Target conformality and heterogeneity as well as target coverage for PTV1 are comparable for both techniques. The target coverage for PTV2 can be significantly improved using carbon ion beams (median 95% coverage 93.7% vs 87%; p = 0.039). Furthermore, the mean doses to the OAR can be reduced by 8.3% (median % reduction of mean doses to OAR; p = 0.00001) using carbon ions. CONCLUSIONS: The combination of photon IMRT with carbon ions improves the target coverage for the boost volume and offers better sparing of OAR close to the PTV2 (gross tumor volume) in comparison with photon IMRT alone. A clinical study has been initiated to evaluate whether these potential advantages translate into clinical benefit.
BACKGROUND: It is more and more doubted that adenoids or tonsils have to be examined histologically in pediatric cases, in which history and clinical signs and symptoms are conclusive for chronic inflammation or hyperplasia. It is unknown whether there is any conformity about this question in Germany. METHOD: The heads of all clinical departments of otorhinolaryngology (n=149) and a similar number of ENT specialists from the Berufsverband Deutscher HNO-Arzte (n=150) were surveyed by a simple questionnaire. They were asked if they would routinely send resected tissue for histopathological examination in the case of a child, up to 10 years old, presenting with a history and a status of chronic inflammation or hyperplasia and undergoing adenoidectomy or tonsillectomy. Furthermore they were asked whether they remembered any unusual or surprising report in this group of patients. RESULTS: Feedback was 79%. All tissue was sent for examination by 59% of the colleagues answering this evaluation; palatine tonsils only were sent for examination by 14%, and 27% of the physicians did not send any tissue for histological work-up. During their professional career 17% of the responding ENT specialists had received a surprising result from the histopathological examination. CONCLUSION: We do not have any consensus practiced in Germany concerning the necessity to send tissue for histopathological examination following adenoidectomy or tonsillectomy in children. It seems helpful to seek such consensus of opinion for medicolegal and socioeconomic reasons.
In a monocenter prospective randomized double-blind clinical trial the efficacy of homeopathic treatment was investigated on children with adenoid vegetations justifying an operation. Patients were treated with either homeopathic remedies such as Nux vomica D200, Okoubaka D3, Tuberculinum D200, Barium jodatum D4 and Barium jodatum D6 or with placebo. The duration of the study for each patient was 3 months. Examination of the ears using a microscope, rhinoscopy, stomatoscopy and pharyngoscopy, as well as tympanometry and audiometry were performed after 4, 8 and 12 weeks. Out of a total of 97 children studied between the ages of 4 to 10 years 82 could be analyzed. At the end of the study no operation was required in 70.7% of the placebo-treated children and in 78.1% of the children treated with homeopathic preparations. These results show no statistical significance.
Adenoid cystic carcinoma (ACC) of the uterine cervix is a rare primary neoplasm of the uterus that occurs in post-menopausal women; its radiological findings have not been described previously. We present the MR findings of a case of ACC. The mass exhibited homogeneous low-signal intensity on T1-weighted images. On T2-weighted images, the mass showed high-signal intensity with a lobulated contour and multiple septum-like internal architectures. It also contained spots of very high-signal intensity, which would represent the mucin in the glandular lumen. The multiple septum-like internal architectures probably represented interglandular fibrous stroma. These MRI findings may be helpful for future diagnoses of ACC of the uterine cervix.
The purpose of this study was to compare the preoperative symptoms of children who had adenoid hypertrophy with postadenoidectomy symptoms. Sixty children undergoing adenoidectomy were included in this prospective uncontrolled study at the Farabi Hospital of Karadeniz Technical University, an academic tertiary medical center. The symptoms of each child were described by their parents. Adenoidectomy with myringotomy alone or with tympanostomy tube placement was performed in all children. Two months after the operation, the children were re-evaluated for remaining or residual symptoms. Nasal obstruction, mouth breathing, snoring, hearing loss and nasal discharge were present preoperatively in 55 (91.6%), 51 (85%), 50 (83%), 28 (46%) and 45 (75%), respectively. We found that 53 of 60 children (88.3%) completely recovered from their preoperative symptoms. Of the remaining seven patients, four had persistent nasal obstruction, five mouth breathing, three snoring and two hearing loss. We also noted that the parents of 53 of 60 children were satisfied after the operation. Adenoidectomy provided significant relief and improvement of preoperative presenting symptoms, and it also showed a high rate of parent satisfaction.
Six distinct alpha(IV) chains in the basement membrane (BM) of adenoid cystic carcinoma (ACC) of the salivary gland were immunohistochemically examined by anti-alpha(IV) chain-specific antibodies, and their expressions were compared with the histological subtypes and the expressions of cytokeratin 19 (CK19), cytokeratin 14 (CK14) and alpha-smooth muscle actin (alpha-SMA) and Ki-67. In the BM of normal salivary ducts, alpha1(IV), alpha2(IV), alpha5(IV) and alpha6(IV) chains were continuously stained, but alpha3(IV) and alpha4(IV) chains were negative. In the tubular and cribriform subtypes of ACC, tubules with continuous staining of alpha5(IV) and alpha6(IV) chains showed the biphasic-staining pattern among the expressions of CK19, CK14 and alpha-SMA. However, in cancer-cell nests with discontinuous or negative staining of alpha5(IV) and alpha6(IV) chains, the biphasic pattern was ambiguous. In the solid subtype, the staining of alpha1(IV) and alpha2(IV) chains was discontinuous, the staining of alpha5(IV) and alpha6(IV) chains was negative and the biphasic-staining pattern was unclear. The mitotic activity of cancer cells analyzed by the Ki-67 labeling index was significantly related to the expression of alpha5(IV) and alpha6(IV) chains in the cribriform subtype. These results suggest that BM irregularity with the differential expression of alpha(IV) chains in ACC closely relates to cell proliferation, cell differentiation and histological structure.
We report a unique case of adenoid cystic carcinoma (ACC) of the maxillary sinus, with gradual histologic transformation from lower-grade ACC (cribriform and tubular types) to high-grade adenocarcinoma (HGA) showing a sequential histologic spectrum via solid-type ACC. A 74-year-old man presented with swelling and mild pain of the right cheek. CT scan showed a mass measuring approximately 4 cm, with marked bone destruction in the right maxillary sinus. A surgically resected specimen revealed that the tumor was comprised of three different components: HGA and solid-type ACC in the central portion and lower-grade ACC in the periphery. The tumor was discriminated from a dedifferentiated carcinoma or hybrid tumor. Autopsy specimens also demonstrated both solid-type ACC and HGA components in the lung and spleen. Immunohistochemically, positive staining of p53 protein was detected on both solid-type ACC and HGA cells, but cyclin D1 and HER2/neu was only seen in HGA cells. Solid-type ACC cells were immunoreactive for CD117 (c-kit), but lower-grade ACC and HGA cells were negative. This case suggests that the overexpression of CD117, p53 protein, cyclin D1, and HER2/neu might be involved in the progression from lower-grade ACC to solid-type ACC and HGA.
Hybrid tumours of the salivary glands are very rare entities composed of two different tumours, each of which conforms with an exactly defined category. We describe an unusual hybrid carcinoma of the palate; it was comprised of an adenoid cystic carcinoma and a salivary duct carcinoma with a transitional region. These two different compartments showed different characteristics as regards cellular differentiation, proliferative activity, and expression of oncogene and tumour suppressor oncogene proteins, as revealed by using markers for muscle actin, keratin, vimentin, S-100 protein, GFAP, Ki-67, p53, and c-erbB-2 proteins. This case is the first reported with overexpression of p53 and c-erbB-2 proteins in the tumour entities. Salivary gland tumours consist of heterogeneous histological groups, and each has morphological diversity. This case indicates that some of the oncogene and tumour suppressor oncogene proteins may help to produce the histological heterogeneity of the salivary gland tumour.
Adenomyoepithelioma is a mixed epithelial and myoepithelial tumour. In rare cases adenomyoepitheliomas give rise to carcinomas with epithelial, myoepithelial, or mixed epithelial and myoepithelial differentiation. Carcinomas arising in adenomyoepithelioma range from low grade to high grade, and 15 cases have been reported in the literature. We describe a 36-year-old woman with a very rare adenoid cystic carcinoma arising in a tubular adenomyoepithelioma. The histogenesis of carcinoma arising in an adenomyoepithelioma is discussed.
The prognosis of salivary gland adenoid cystic carcinoma (ACC) depends on the clinical stage, the location of the primary tumor, and the histologic growth pattern. ACCs with a cribriform growth pattern have a better prognosis than those with a solid growth pattern; however, clear-cut grading criteria have not yet been established, and therefore prognostic indicators on a molecular level are of special interest. In order to analyze tumor tissue with different growth patterns, cribriform and solid tumor areas of 25 patients were microdissected and separately analyzed for loss of heterozygosity (LOH) at nine polymorphic microsatellite markers located between 6q14 and 6q27. LOH was detected in 19/25 (76%) patients and LOH rates were highest at markers D6S441, D6S310, D6S311 and UTRN, which are located at 6q23-25. Combined analysis of LOH at these four markers shows that in primary tumor subtype foci with cribriform growth pattern LOH is associated with high TNM stages (P<0.01), high T stages (P=0.01), positive lymph node status (P=0.03), an unfavorable disease course (P=0.02), and the presence of >10% solid growth pattern (P=0.05). In contrast, in primary tumor subtype foci with solid growth pattern, no significant differences in LOH rates were found in patients from prognostically and histologically favorable versus unfavorable patient groups. The frequent occurrence of LOH at 6q23-25 and the correlation of LOH rates with prognostic parameters indicate that a prognostically important tumor suppressor gene is located in this chromosomal area.
PURPOSE: Adenoid cystic carcinoma (ACC) is one of the most common malignant tumours of the salivary glands characterized by multiple recurrences and distant metastasis resulting in significantly worsening prognosis. Galectin-3, a member of the beta-galactoside-binding lectin family, has been implicated in tumour progression, metastasis, and found to have prognostic value. The aim of the study was to determine galectin-3 expression in ACC and correlate it with clinicopathological features and patient survival. METHODS: Galectin-3 expression was investigated in paraffin sections of 35 ACC of the head and neck. Patients were divided into two groups based on a threshold of 5% positivity in the tumour cell population. The mean follow-up period for all patients was 90.1 months (range 3-300.1 months). RESULTS: Seventeen (48.6%) tumour specimens were considered galectin-3-positive. Galectin-3 reactivity was significantly associated with regional and distant metastasis (P=0.045 and P<0.001, respectively). There was no statistical significance in the correlation of galectin-3 expression and disease-free survival and overall survival rate (P=0.095 and 0.102, respectively). CONCLUSION: Galectin-3 may be used as an indicator in the prediction of metastatic spread in ACC.
Adenoid cystic carcinoma of the breast is an uncommon form of cancer, and only a few articles have described the cytological findings of this disease. We report herein the case of a 48-year-old woman who presented with a breast mass beneath the nipple, the aspirate from which consisted of globules of mucous balls surrounded by epithelial cells with scant cytoplasm and hyperchromatic nuclei. Microscopically, the tumor was formed by myoepithelial cells and glandular epithelial cells in a biphasic pattern. Immunohistochemical study revealed positivity for smooth muscle actin. A left total mastectomy with axillary lymph node dissection was performed. None of the 22 axillary lymph nodes contained metastases, and the patient remains well and free from recurrence 29 months after her operation. This case report provides some information about the cytological diagnosis and the accuracy of fine-needle aspiration, which must be considered despite the rarity of this disease.
Adenoid cystic carcinoma (ACC) is a relatively common head and neck tumor which grows slowly but locally aggressive and prone to recurrence. ACC most commonly arises in the major and minor salivary glands. Some rare cases of intracranial ACC with an unknown primary site have been reported. The authors present the first case of primary intracranial ACC of the suprasellar region. A 34 year-old female presented with one month's duration of visual disturbance and galactorrhea. Magnetic resonance image (MRI) showed intra- and suprasellar mass mimicking a craniopharyngioma. There was no evidence of invasion from an extracranial site. The operative findings showed the mass existed under the arachnoid membrane and seemed to originate from the pituitary stalk. Pathological diagnosis was ACC. After the first operation, local recurrence and CSF dissemination to the lower clivus occurred within two months. Recurrence repeatedly treated by Gamma knife radiosurgery 10 times and 4 times by surgery during a 3 year follow-up period. But total removal of the tumor at the first operation along with radiosurgery may control the lesion and prevent further recurrence.