Adenoid Cystic Carcinoma Of Lacrimal Gland Research Articles

Whole Exome Sequencing of Lacrimal Gland Adenoid Cystic Carcinoma.

PurposeTo identify genomic mutations in lacrimal gland adenoid cystic carcinoma (LGACC) samples from patients.MethodsGenomic DNA was extracted from LGACC specimens. Whole exome sequencing (exome-seq) was conducted to screen for mutations. Capillary sequencing was performed to verify mutations in genes shared by multiple samples. Luciferase assays were used to evaluate functional consequences of NOTCH1 mutations.ResultsThe mutation profile of LGACC was complicated. The most frequently mutated gene observed (28.6%) was bromodomain PHD finger transcription factor (BPTF). No mutation was identified in common cancer genes such as TP53, KRAS, and BRAF. However, mutations predicted to be functionally severe were accumulated in the Notch signaling pathway including NOTCH1 and NOTCH2, of which mutations have been reported in head/neck adenoid cystic carcinoma (ACC). Of 14 LGACC samples, five samples carry mutations in Notch pathway genes. Capillary sequencing verified all the mutations in the two NOTCH genes identified by exome-seq. Compared to the wild-type NOTCH1, three frame shifting mutations and two missense mutations (C387W and L1600Q) increased luciferase activity approximately 10- to 25-fold.ConclusionsMajor genomic mutation profiles in LGACC were uncovered by exome-seq. Although preliminary in nature, the Notch pathway could be a potential therapeutic target for LGACC.

Current treatment of lacrimal gland carcinoma.

The traditional treatment for lacrimal gland carcinoma is orbital exenteration followed by radiation therapy. However, orbital exenteration does not prevent distant relapse and death, and some patients experience local-regional recurrence after exenteration. More recently, eye-sparing surgery and adjuvant radiation therapy and chemotherapy have gained popularity in the treatment of lacrimal gland carcinoma. Preliminary studies show that these approaches are associated with reasonable local control rates and ocular toxicity profiles. In this review, we discuss recent studies of treatment of lacrimal gland carcinoma, including studies of potential molecular treatment targets. Recent studies suggest promising results for neoadjuvant intra-arterial chemotherapy followed by orbital exenteration and adjuvant intravenous chemotherapy, but only in patients with an 'intact lacrimal artery'. Recent studies of globe-sparing surgery followed by adjuvant proton radiation therapy or concurrent chemoradiation suggest good local control, reasonable rates of eye preservation, and low risk of serious ocular toxic effects. Larger tumor size and predominant basaloid histology seem to be associated with higher risk of local-regional recurrence and distant metastasis. One study showed oncogenic mutations in more than half of cases of lacrimal gland adenoid cystic carcinoma, with KRAS mutations in 10 of 24 patients, suggesting potential benefit of treatments targeting the EGFR-RAS-RAF cascade. In selected patients with lacrimal gland carcinoma, eye-sparing surgery with eye-sparing radiation therapy offers preservation of visual function with good local control and minimal radiation-induced ocular toxic effects. In patients with recurrent or metastatic disease, a search for actionable cancer-associated mutations may be prudent.

The value of conventional and contrast-enhanced ultrasound in the diagnosis of tumors in front of the orbit equator

Objective To explore the value of conventional and contrast-enhanced ultrasound(CEUS) in the diagnosis of tumors in front of the orbit equator. Methods Clinical data included conventional ultrasound and CEUS image of 133 cases (156 eyes) with orbital lesions were analyzed retrospectively. All lesions were confirmed by surgical pathology or clinical cure, which included 26 cases of cystic lesions (epidermoid cysts, dermoid cysts, sebaceous cysts and mucous cysts), 29 cases (30 eyes) of orbital hemangioma, 13 cases (16 eyes) of inflammatory pseudotumor, 11 cases of lacrimal gland pleomorphic adenoma, 3 cases (5 eyes) of lymphoepithelial lesion, 36 cases (53 eyes) of malignant lymphoma, 5 cases of meibomian gland carcinoma, 4 cases of lacrimal gland adenoid cystic carcinoma, 3 cases of malignant melanoma and 3 cases of basal cell carcinoma. Results CEUS of benign cystic lesions showed the tumors were enhanced inhomogenously. Most CEUS of benign tumours displayedslow in and slow out, and high enhancement when reached the peak but the size of images had no change. While CEUS of malignant tumours showedfast in and fast out, homogeneous or inhomogeneous high enhancement and ill-defined or irregular shape, and volume increased compared to two-dimensional ultrasound when reached the peak.Some other former district orbital lesions, such as cystic lesion, cavernous hemangioma, pleomorphic adenoma and malignant lymphoma, had their own ultrasound features. Conclusions CEUS combines with conventional ultrasound may contribute to the qualitative diagnosis of tumors in front of the orbit equator. Key words: Contrast-enhanced ultrasound; Orbital neoplasms

The cultivation and identification of lacrimal gland adenoid cystic cancer stem cells

To isolate and cultivate the Lacrimal gland Adenoid Cystic Carcinoma cells line, study Cancer Stem Cells properties. Experimental study. Lacrimal gland adenoid cystic carcinoma cancer stem cells were cultivated in serum-free suspension culture and the morphological changes were observed. Cells were divided into two groups, the LACC-CSC experimental group and the LACC control group. The flow cytometry instrument was used to detect the expression of classical stem cell markers CD133 and ABCG2. Transwell chamber was used to detect the cancer stem cell aggressivity and differentiated into the vascular endothelial cells. The tumorigenic force in vitro xenotransplantation were applied. LACC cells can grow suspensively after vaccinated in serum free medium and form tumor microspheres after 10-12 days. Flow cytometry experiments showed that the expression ratio of stem cell markers CD133 in LACC-CSC was (35.67 ± 6.86)%, significantly different to LACC with (0.46 ± 0.48)%, (t = 8.867, P < 0.05). Similarly, the expression ratio of stem cell marker ABCG2 in LACC-CSC was (39.99 ± 4.54)%, significantly different to LACC with (6.75 ± 1.34)%, (t = -9.932, P < 0.05). In vitro experiment of Matrigel invasion, LACC-CSC went through the matrigel basement membrane averagely (32.60 ± 8.79)/HP contrary to LACC with average (10.20 ± 2.77)/HP after 24 hours, showing statistically significance (t = 5.433, P < 0.05) between the two groups. After training for 48 hours, the difference between two groups was still obvious (t = 5.779, P < 0.05) with LACC-CSC average (62.60 ± 4.83)/HP to LACC (44.00 ± 5.34)/HP. When induced by serum medium containing VEGF and bFGF, LACC-CSC grew adherent gradually and cell morphological changes occurred after continuous induction to long spindle cells. When cultured into three-dimensional matrix structure they formed vessel samples and expressed vascular endothelial marker CD31 and CD34. Transplanted tumor in vitro experiment, mice of LACC-CSC group grew tumors in 9 days with 100% tumorigenic rate, whereas LACC group 12 days with 100% tumorigenic rate. LACC-CSC can be obtained through serum-free culture method. LACC-CSC grew suspensively and expressed classical stem cell markers. LACC-CSC were identified as cancer stem cells with stronger migration and invasion. LACC-CSC have tumorigenic force and multi-directional differentiation potential with general characteristics of the stem cell.

Expression of metabolism-related proteins in lacrimal gland adenoid cystic carcinoma.

To investigate the expression and the clinical implications of metabolism-related proteins in lacrimal gland adenoid cystic carcinoma (ACC) in comparison with salivary gland ACC. Human tissue samples of lacrimal gland ACC (n = 11) and salivary gland ACC (n = 64) were analyzed. Immunochemistry was used to measure expression of proteins related to glycolysis (glucose transporter 1, hexokinase II, carbonic anhydrase IX, and monocarboxylate transporter 4 [MCT4]), glutaminolysis (glutaminase 1 [GLS1], glutamate dehydrogenase [GDH], and amino acid transporter 2 [ASCT2]), mitochondria (adenosine triphosphate [ATP] synthase, succinate dehydrogenase A [SDHA], and succinate dehydrogenase B), and glycolytic intermediate metabolism (phosphoserine phosphatase [PSPH], serine hydroxymethyl transferase 1 [SHMT1]). GLS1 and ASCT2 were more highly expressed, and GDH, ATP synthase, and SDHA were expressed to a lesser degree in lacrimal gland ACC than in salivary gland ACC (P < .05). Lacrimal gland ACC showed less of a mitochondrial phenotype than did salivary gland ACC (P = .001). Positivity of MCT4 and PSPH was related to shorter disease-free survival, and SHMT1 was related to shorter overall survival (P < .05). Lacrimal gland ACC exhibited higher expression of GLS1 and ASCT2, compared with salivary gland ACC. Overexpression of MCT4, PSPH, and SHMT1 was associated with poorer prognosis.

Adjuvant Radiotherapy with Three-Dimensional Conformal Radiotherapy of Lacrimal Gland Adenoid Cystic Carcinoma.

Adenoid cystic carcinoma (ACC) of lacrimal gland is a rare tumour with aggressive behaviour. There is sparse data to address optimum therapy for such tumours. So, the present study was aimed at evaluating the role of adjuvant three dimensional conformal radiotherapy (3D-CRT) in cases of incomplete (R1) resection along with review of literature pertaining to management of lacrimal adenoid cystic carcinoma. We retrospectively reviewed the demographic and treatment data of 10 biopsy proven ACC of lacrimal gland patients, treated from December 2006 to June 2013. They were treated with radiotherapy following surgical resection. Eight patients underwent gross total excision of the tumour mass (enbloc excision) followed by conformal radiotherapy to a dose of 60 Gray/30fractions/ 6 weeks. Two patients with advanced disease were treated with palliative radiotherapy after biopsy. The median age was 32 years. There were equal numbers of male and female patients. The median duration of symptoms was 7 months. At a median follow up of 21 months, eight patients had no evidence of disease and had complete tumour response, two patients worsened, and one of the two had systemic failure with bone metastasis. Despite a small sample size and short follow, enbloc surgical excision with adjuvant radiotherapy is well tolerated and shows good control in ACC of lacrimal gland.

Influence of ribosomal protein L39-L in the drug resistance mechanisms of lacrimal gland adenoid cystic carcinoma cells.

Cancer constitutes a key pressure on public health regardless of the economy state in different countries. As a kind of highly malignant epithelial tumor, lacrimal gland adenoid cystic carcinoma can occur in any part of the body, such as salivary gland, submandibular gland, trachea, lung, breast, skin and lacrimal gland. Chemotherapy is one of the key treatment techniques, but drug resistance, especially MDR, seriously blunts its effects. As an element of the 60S large ribosomal subunit, the ribosomal protein L39-L gene appears to be documented specifically in the human testis and many human cancer samples of different origins. Total RNA of cultured drug-resistant and susceptible lacrimal gland adenoid cystic carcinoma cells was seperated, and real time quantitative RT-PCR were used to reveal transcription differences between amycin resistant and susceptible strains of lacrimal gland adenoid cystic carcinoma cells. Viability assays were used to present the amycin resistance difference in a RPL39-L transfected lacrimal gland adenoid cystic carcinoma cell line as compared to control vector and null-transfected lacrimal gland adenoid cystic carcinoma cell lines. The ribosomal protein L39-L transcription level was 6.5-fold higher in the drug-resistant human lacrimal gland adenoid cystic carcinoma cell line than in the susceptible cell line by quantitative RT-PCR analysis. The ribosomal protein L39-L transfected cells revealed enhanced drug resistance compared to plasmid vector-transfected or null-transfected cells as determined by methyl tritiated thymidine (3H-TdR) incorporation. The ribosomal protein L39-L gene could possibly have influence on the drug resistance mechanism of lacrimal gland adenoid cystic carcinoma cells.

Recurrent orbital space-occupying lesions: a clinicopathologic study of 253 cases.

To analyze the clinical features, histopathologic classification and frequencies of various types of recurrent orbital space-occupying lesions. A retrospective study was carried out in 253 consecutive patients with recurrent orbital space-occupying lesions treated by surgical excision in the Institute of Orbital Diseases, the General Hospital of the Armed Police Force from January 2009 to December 2010. The patients included 123 males and 130 females aged 2 to 78 years (mean, 36.2 years), and the last recurrence interval after operation ranged from 1 month to 40 years (median, 4.75 years). Of all the cases, 159 (62.8%), 65 (25.7%), 20 (7.9%), 8 (3.2%) and 1 (0.4%) had previously experienced once, twice, three, four and six times of surgeries, respectively. Among them, 29 (11.5%) cases had recurred 3 times or over, and 37 (14.6%) cases got recurrence in 10 or more years postoperatively. Most of the patients with local recurrence presented with various clinical manifestations, while 31 (12.3%) cases were symptom-free. Two hundred and thirty-one (91.3%) cases underwent surgical removal of the recurrent orbital lesions, and another 22 (8.7%) cases had to receive the exenteration of orbit. Categories of these recurrent orbital lesions after operation were as follows: lacrimal gland tumors, 65 (25.7%) cases; vasogenic diseases, 54 (21.3%) cases; neurogenic tumors, 42 (16.6%) cases; secondary tumors, 24 (9.5%) cases; orbital inflammation, 21 (8.3%) cases; myogenic tumors, 14 (5.5%) cases; fibrous and adipose tumors, 12 (4.7%) cases; lympho-hematopoietic tumors, 7 (2.8%) cases; bone or cartilage tumors, 7 (2.8%) cases; orbital cysts, 6 (2.4%) cases; and indefinitely differentiated tumor, 1 (0.4%) case. The 10 top histopathologic diagnoses were lacrimal gland pleomorphic adenoma, hemangiolymphangioma, lacrimal gland adenoid cystic carcinoma, meningioma, inflammatory pseudotumor, neurofibroma, sebaceous gland carcinoma, vascular malformation, rhabdomyosarcoma and hemangioma. The variety of recurrent orbital lesions after operation includes mainly of tumors except for vascular malformation and orbital inflammatory lesions. The lacrimal gland epithelial tumor is most prone to relapse after resection, and early and longer-term postoperative follow-up is needed.

Neutron Radiotherapy for Adenoid Cystic Carcinoma of the Lacrimal Gland

Lacrimal gland adenoid cystic carcinomas are rare, aggressive orbital tumors that share histopathologic similarities with salivary gland malignancies. Neutron radiotherapy may be useful for treatment due to its high biological effectiveness for salivary malignancies. The authors retrospectively reviewed the outcomes for 11 lacrimal gland adenoid cystic carcinoma patients treated with neutrons from 1988 to 2011. Most had undergone surgery prior to radiation therapy. However, gross residual disease was present in 8 patients. The most common American Joint Committee on Cancer stage was T4cN0M0. Four patients with skull base involvement received a radiosurgery boost and 1 received a proton therapy boost. Median follow up was 6.2 years. Median overall survival was 11.1 years and median disease-free survival was 6.3 years. Five-year local control was estimated by the Kaplan-Meier method as 80%. Three patients had a local recurrence; 4 developed distant metastases. Six patients died. Seven patients had intact vision in the affected eye before neutron radiation. Two required enucleation for a painful dry eye. Of the 5 who avoided an enucleation, 3 had either severe visual impairment (20/400) or only light perception and 2 were without known vision compromise or complications at the time of their death. One patient developed asymptomatic frontal lobe radionecrosis after 2 courses of radiation therapy. Neutron radiation therapy achieved excellent 5-year local control in this series of high-risk patients, with most cases having gross residual disease. Late recurrences and distant metastases remain a challenge. Meaningful ipsilateral vision preservation was not possible in most cases in the long term, although only 2 patients required an enucleation for treatment effects.

Adenoid cystic carcinoma of the lacrimal gland: role of nuclear survivin (BIRC5) as a prognostic marker

This study aimed to evaluate the expression of nuclear survivin in adenoid cystic carcinoma (ACC) of the lacrimal gland and to determine if this expression is associated with histopathological features, markers of apoptosis and proliferation or clinical outcomes. Immunohistochemical staining for survivin, p53, Ki-67 and Bcl-2 was analyzed in 55 cases of ACC of lacrimal gland. Thirty-one cases (56.3%) expressed nuclear survivin. All cases expressed p53, Ki-67 and Bcl-2. Eleven cases (35.5%) had a high nuclear survivin score (NS-SCORE) and 20 cases (64.5%) had a low NS-SCORE. Cases with a high NS-SCORE had a shorter progression-free survival (PFS) (P < 0.0001), higher expression of Ki-67 (P < 0.005) and a solid tumour pattern >30% (P < 0.005). Nuclear expression of survivin impacts prognosis significantly and is thus a promising prognostic marker in ACC of the lacrimal gland.

Long-Term Outcomes of Neoadjuvant Intra-arterial Cytoreductive Chemotherapy for Lacrimal Gland Adenoid Cystic Carcinoma

To compare the long-term outcomes after intra-arterial cytoreductive chemotherapy (IACC) with conventional treatment for lacrimal gland adenoid cystic carcinoma (ACC). Retrospective case series. Nineteen consecutive patients treated with IACC, followed by orbital exenteration, chemoradiotherapy, and intravenous chemotherapy. Analyses of the histologic characteristics of biopsy specimens, extent of disease at the time of diagnosis, diagnostic surgical procedures, incidence of locoregional recurrences or distant metastases, disease-free survival time, response to IACC, tumor margins at definitive surgery, and toxicity and complications. Disease relapse, disease-free survival, and chemotherapeutic complications. Eight patients with an intact lacrimal artery had significantly better outcomes for survival (100% vs. 28.6% at 10 years), cause-specific mortality, and recurrences (all P = 0.002, log-rank test) than conventionally treated patients from the University of Miami Miller School of Medicine. These 8 patients (group 1) had cumulative 10-year disease-free survival of 100% compared with 50% for 11 patients (group 2) who had an absence of the lacrimal artery or deviated from the treatment protocol (P = 0.035) and 14.3% for conventionally treated patients (P<0.001). Likewise, group 2 was associated with lower cause-specific mortality than the institutional comparator group (P = 0.038). Prior tumor resection with lateral wall osteotomy, delay in IACC implementation or exenteration, and failure to adhere to protocol are risk factors for suboptimal outcomes. Neoadjuvant IACC seems to improve overall survival and decrease disease recurrence. An intact lacrimal artery, no disruption of bone barrier or tumor manipulation other than incisional biopsy, and protocol compliance are factors responsible for favorable outcomes. The chemotoxicity complication rate is limited and manageable.

Imaging feature and clinical histological analysis of 12 adenoid cystic carcinoma of the lacrimal gland

To study the imaging and histologic features of adenoid cystic carcinoma (ACC) of the lacrimal gland. It was a retrospective case series study. Twelve patients with ACC of the lacrimal gland were surgically treated in Tianjin First Center Hospital from September 2009 to November 2011. The 5 men and 7 women aged from 22 to 63 years (average 42.6 years). The imaging and histologic features of 12 cases with ACC of the lacrimal gland pathologically confirmed were retrospectively reviewed. Twelve cases were performed with CT scan, 10 cases with MRI scan. All patients were followed up by telephone and reexamination in the outpatient service, and 2 patients were lost to follow-up. The lesions originated in the orbital lobe of the lacrimal gland. The most common presenting symptom was pain; it was followed by proptosis, ptosis, decreased visual acuity and diplopia. The 7th Edition of the American Joint Committee on Cancer TNM Classification system for Lacrimal Gland Tumors stages were as follows: T1N0M0 1 patients, T2N0M0 4 patients, T4N0M0 7 patients. Preoperative CT imaging suggested that bony involvement of the lacrimal gland fossa in 7 patients;this was histologically confirmed in 6 of the 7. Preoperative CT imaging suggested no bone involvement in 5 patients, 1 of whom had bone involvement by histology. The positive rate of CT scan was 6/7. Preoperative MRI imaging suggested that bony involvement of the lacrimal gland fossa in 7 patients;this was histologically confirmed in 6 of the 7. Preoperative MRI imaging suggested no bone involvement in 3 patients who were confirmed by histology. The positive rate of MRI scan was 6/7. Overall, 7 of 12 histologically evaluable cases had bone invasion. Five of the histologically proven 7 patients with bone involvement had a predominantly basaloid pattern, 2 mixed pattern. Three patients had local recurrence. The imaging for ACC of the lacrimal gland is characteristic. ACC of the lacrimal gland is associated with the high rate of bone invasion. The basaloid variety has more aggressive biologic behavior. The risk of local recurrence may be associated with the histologic types and stages of lacrimal gland adenoid cystic carcinoma.

Multiple cutaneous metastases: a rare and late sequelae of lacrimal gland adenoid cystic carcinoma

Primary cutaneous adenoid cystic carcinoma (ACC) is an uncommon malignancy with approximately 60 cases reported in the literature. Metastatic ACC to the skin is even less frequent with eight previously published cases, including only one arising from the lacrimal gland. We report a case of a 61-year-old male, who developed multiple cutaneous metastases to the scalp and back from a lacrimal gland ACC 21 years after the initial diagnosis. Initially, in the absence of the past clinical history, the cutaneous lesions were interpreted as primary ACC of the skin. Histopathologically, the metastatic lesions were poorly circumscribed dermal and subcutaneous nodules that were composed of islands of basaloid epithelial cells with ducts and cribriform pattern, mucinous stroma and prominent perineural invasion. Histomorphologically, the cutaneous tumors were similar to the primary ACC. The cutaneous metastases were preceded by tumor dissemination to the lung and other visceral organs, and ultimately led to patient's demise. This report illustrates the importance of juditious clinico-pathologic correlation and raises awareness regarding prolonged latency of cutaneous metastases in patients with visceral ACC.

Influence of Ginkgo biloba extract on the proliferation, apoptosis of ACC-2 cell and Survivin gene expression in adenoid cystic carcinoma of lacrimal gland

ObjectiveTo explore the influence of extract of Ginkgo biloba (EGB) on the proliferation, apoptosis of ACC-2 cell and Survivin gene expression in adenoid cystic carcinoma (ACC) of lacrimal gland. MethodsACC-2 cell in human with ACC of lacrimal gland was in vitro cultured. MTT method was used for cell proliferation detection. Annexin V/PI double-staining flow cytometer was used to detect cell apoptosis and cell cycle. Survivin gene expression was analyzed by RT-PCR and Western blotting. ResultsEGB had inhibitory effect on the proliferation of ACC-2 cell with significant dose-effect relationship, and there was statistical difference when compared with the control group (P<0.01). The inhibitory concentration 50 % (IC50) is 88 mg/L. The flow cytometer test indicated that EGB can gradually increase ACC-2 cell in G0-G1 stage and decrease it in G2-M and S stage. With the increase of dose, the apoptosis rate of ACC-2 cell was obviously increased (P<0.05 or P<0.01). EGB had certain inhibitory effect on Survivin gene expression of ACC-2 cell, and Survivin gene expression was decreased with the increasing of the EGB concentration (P<0.01). ConclusionsEGB can effectively inhibit Survivin gene expression of ACC-2 cell in human with ACC of lacrimal gland, induce the apoptosis of ACC-2 cell and inhibit tumor cell proliferation.

Lacrimal Gland Adenoid Cystic Carcinoma with High-Grade Transformation to Myoepithelial Carcinoma: Report of a Case and Review of Literature

Adenoid cystic carcinoma (AdCC) is the most frequent malignant neoplasm of the lacrimal glands. Like its salivary gland counterpart, lacrimal AdCC can rarely undergo high-grade transformation ("dedifferentiation"). We herein report the clinical, radiographic and microscopic findings of a lacrimal gland AdCC with high grade transformation, occurring in a 39-year-old female patient. In contrast to salivary gland AdCC with high-grade transformation, which usually shows a high grade component with "ductal" differentiation, in the case presented, the "dedifferentiated" component showed morphologic and immunohistochemical features of myoepithelial differentiation.

Adenoid cystic carcinoma of the lacrimal gland: CT and MRI findings

To highlight the computed tomography (CT) and magnetic resonance imaging (MRI) characteristics of lacrimal gland adenoid cystic carcinoma. We reviewed the clinical records and imaging findings of 17 patients who were histopathologically diagnosed with primary adenoid cystic carcinoma of the lacrimal gland between August 1998 and November 2010. Orbital CT scans were performed in 17 patients and MRI scans were obtained in 13 patients. On the CT scans, out of 4 cases of adenoid cystic carcinoma, 3 showed characteristic features of a soft tissue mass with poorly enhancing areas representing cystic changes, and 4 of 17 cases had contiguous bony erosion. On the MRI scans, all cases had mixed signal intensity on T2-weighted images, and 6 cases showed poorly enhancing areas. Enhanced CT and MRI (including T2-weighted) are helpful in the diagnosis of primary adenoid cystic carcinoma of the lacrimal gland.

Bone Invasion by Adenoid Cystic Carcinoma of the Lacrimal Gland: Preoperative Imaging Assessment and Surgical Considerations

To identify the incidence of radiologically and histologically documented bony invasion of the lacrimal gland fossa by adenoid cystic carcinoma. The authors reviewed the records of all 18 patients with lacrimal gland adenoid cystic carcinoma surgically treated at their institution from 1997 to 2009 for imaging findings (blinded review) and histologic findings on evaluation of the lacrimal gland fossa. Preoperative CT and/or MRI findings were available for 17 patients. The 8 men and 10 women ranged in age from 9 to 69 years. American Joint Committee on Cancer tumor stages after preoperative imaging were as follows: T1N0M0, 2 patients; T2N0M0, 5 patients; T3aN0M0, 2 patients; T3bN0M0, 5 patients; T3bN0M1, 2 patients; T4bN0M0, one patient; and TxN0M0, one patient. Preoperative imaging suggested bony involvement of the lacrimal gland fossa in 13 patients (76.5%); this was histologically confirmed in 11 of the 13. Preoperative imaging suggested no bone involvement in 4 patients, 3 of whom had bone involvement by histology. Overall, 14 of 17 histologically evaluable cases (82.3%) had invasion of the lacrimal gland fossa. Histologic findings of bone/periosteal involvement led to upstaging of 3 tumors. Metastases developed in 8 of 18 patients and trended with basaloid histology (p = 0.066). Adenoid cystic carcinoma of the lacrimal gland is associated with bone invasion in essentially all but the smallest of tumors (T1). This high rate of bone involvement may warrant addressing the bony walls during surgery for adenoid cystic carcinoma of the lacrimal gland.

Adjuvant chemotherapy in lacrimal gland adenoid cystic carcinoma

Pediatric Blood & CancerVolume 53, Issue 6 p. 1163-1164 Letter to the Editor Adjuvant chemotherapy in lacrimal gland adenoid cystic carcinoma Rachna Meel MS, Rachna Meel MS Department of Oculoplastics and Ocular Oncology Service, Dr. Rajendra Prasad Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this authorNeelam Pushker MD, Neelam Pushker MD Department of Oculoplastics and Ocular Oncology Service, Dr. Rajendra Prasad Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this authorSameer Bakhshi MD, Corresponding Author Sameer Bakhshi MD sambakh@hotmail.com Medical Oncology, Dr. B.R.A. Institute Rotary Cancer Hospital, All India Institute of Medical Sciences, New Delhi, IndiaAssociate Professor of Pediatric Oncology, Department of Medical Oncology, Dr. B.R.A. Institute Rotary Cancer Hospital, All India Institute of Medical Sciences, New Delhi, India.===Search for more papers by this author Rachna Meel MS, Rachna Meel MS Department of Oculoplastics and Ocular Oncology Service, Dr. Rajendra Prasad Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this authorNeelam Pushker MD, Neelam Pushker MD Department of Oculoplastics and Ocular Oncology Service, Dr. Rajendra Prasad Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this authorSameer Bakhshi MD, Corresponding Author Sameer Bakhshi MD sambakh@hotmail.com Medical Oncology, Dr. B.R.A. Institute Rotary Cancer Hospital, All India Institute of Medical Sciences, New Delhi, IndiaAssociate Professor of Pediatric Oncology, Department of Medical Oncology, Dr. B.R.A. Institute Rotary Cancer Hospital, All India Institute of Medical Sciences, New Delhi, India.===Search for more papers by this author First published: 17 July 2009 https://doi.org/10.1002/pbc.22175Citations: 6Read the full textAboutPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL Share a linkShare onFacebookTwitterLinked InRedditWechat No abstract is available for this article.Citing Literature Volume53, Issue61 December 2009Pages 1163-1164 RelatedInformation

American Joint Committee on Cancer Classification Predicts Outcome of Patients with Lacrimal Gland Adenoid Cystic Carcinoma

To investigate whether American Joint Committee on Cancer (AJCC) classification at initial diagnosis of lacrimal gland adenoid cystic carcinoma predicts outcome of treatment on local recurrence. Retrospective chart review. Consecutive patients with adenoid cystic carcinoma of the lacrimal gland treated at 8 institutions between January 1986 and December 2007. Clinical records, including pathology reports and imaging studies, were reviewed. AJCC classification, histologic subtype, local recurrence rate, and survival. AJCC classification at initial diagnosis was assessable for 53 patients and was as follows: T1N0M0, 7 patients; T2N0M0, 8 patients; T3aN0M0, 14 patients; T3aNxM0, 1 patient; T3aN0M1, 1 patient; T3bN0M0, 13 patients; T3bN0M1, 1 patient; T4aN0M0, 2 patients; T4bN0M0, 4 patients; T4bN0M1, 1 patient; and T4bNXM0, 1 patient. Thirty-eight (72%) of the 53 patients had >T3 tumors at presentation. Of the 38 patients with >T3 tumors, 20 were treated with orbital exenteration and postoperative adjuvant radiotherapy (RT), 6 were treated with orbital exenteration without RT, and 12 were treated with globe-preserving surgery (10 with RT and 2 without RT). Of the 15 patients with <T3 tumors, 6 were treated with globe-preserving surgery and RT, 2 were treated with globe-preserving surgery without RT, 6 were treated with orbital exenteration with bone removal and RT, and 1 was treated with orbital exenteration with bone removal without RT. Only 1 patient with a <T3 tumor, had local recurrence. Among patients with >T3 tumors, the risk of local recurrence (in the orbit or skull base) was higher in patients treated with conservative surgery as opposed to orbital exenteration and RT. Only 4 (20%) of the 20 patients treated with orbital exenteration and RT had local recurrence, compared with 3 (50%) of the 6 patients treated with orbital exenteration without RT and 8 (67%) of the 12 patients treated with globe-preserving surgery. Overall, 17 (45%) of the 38 patients with >T3 tumors and only 1 (7%) of the 15 patients with <T3 tumors died of disease during the study period. In patients with lacrimal gland adenoid cystic carcinoma, AJCC >T3 disease at initial diagnosis correlates with worse outcomes than does AJCC <T3 disease. The author(s) have no proprietary or commercial interest in any materials discussed in this article.

Clinical Analysis of the Effect of Intraarterial Cytoreductive Chemotherapy in the Treatment of Lacrimal Gland Adenoid Cystic Carcinoma

Clinical Analysis of the Effect of Intraarterial Cytoreductive Chemotherapy in the Treatment of Lacrimal Gland Adenoid Cystic Carcinoma