Treatment of lacrimal gland adenoid cystic carcinoma:a systematic review and Meta-analysis

AIM:To evaluate lacrimal gland adenoid cystic carcinoma(LGACC)of prognosis in patients who underwent different treatment regimens.METHODS:We searched PubMed,EMBASE,and the Cochrane Library for studies done on the treatment of LGACC,between January 1987 and April 2022.A Metaanalysis was conducted to pool the 5-year overall survival rate(OR),and the 5-year recurrence rate(RR)and 5-year metastasis rate(MR)were assessed.RESULTS:The 30 studies involved 585 patients were included in the Meta-analysis.The pooled 5-year OR with surgery alone was 50%,the 5-year RR was 63%,and the 5-year MR was 34%.The pooled 5-year OR with surgery and adjuvant radiotherapy combined was 67%(95%CI 61%,73%),the 5-year RR was 41%,and the 5-year MR was 35%.The pooled 5-year OR with surgery and adjuvant chemoradiotherapy combined was 72%(95%CI 59%,84%),the 5-year RR was 48%,and the 5-year MR was 36%.The pooled 5-year OR with surgery,intra-arterial cytoreductive chemotherapy,and adjuvant chemoradiotherapy combined was 78%(95%CI 68%,89%),the 5-year RR was 15%,and the 5-year MR was 27%.CONCLUSION:Comprehensive treatment is more effective than surgery alone.Surgery combined with intraarterial chemotherapy and adjuvant chemoradiotherapy seems to add value to the therapeutic effect of comprehensive treatment of LGACC but further high-quality research is required to validate this.

Prognostic factors for lacrimal gland adenoid cystic carcinoma:a retrospective study in Chinese patients

AIM:To explore the prognostic factors for lacrimal gland adenoid cystic carcinoma(LGACC)in Chinese patients.METHODS:Clinical and histopathological data were reviewed in patients with pathologically confirmed LGACC.Local recurrence,metastasis,and disease-specific death were the main outcome measures.Univariate and multivariate analyses were performed by the Kaplan-Meier method and a Cox proportional hazard model.RESULTS:This retrospective cohort study included 45 patients with pathologically confirmed LGACC between January 2008 and June 2022.Tumor(T)classification(P=0.005),nodal metastasis(N)classification(P=0.018)and positive margin(P=0.008)were independent risk factors of recurrence;T(P=0.013)and N(P=0.003)classification and the basaloid tumor type(P=0.032)were independent risk factors for metastasis;T classification(P<0.001)was an independent factor of death of disease.In the further analysis,the durations from first surgery to radiotherapy is correlated with metastatic risk in LGACC patients with basaloid component(P=0.022).CONCLUSION:Histological subtype should be emphasized when evaluating prognosis and guiding treatment.Timely radiotherapy may reduce the risk of metastasis in patients with basaloid component.

Optimizing surgical approaches for lacrimal gland adenoid cystic carcinoma to minimize cross-organ invasion

AIM:To evaluate the outcomes of eye-sparing surgery for lacrimal gland adenoid cystic carcinoma and the impact on tumor recurrence and orbital integrity.METHODS:The study enrolled four patients with recurrent lacrimal gland adenoid cystic carcinoma.The outcome focused on the relevance of the integrity of the lateral orbital wall to the occurrence of extraorbital metastasis in the local recurrence of lacrimal gland adenoid cystic carcinoma.RESULTS:Three patients underwent eye-sparing surgery via lateral orbitotomy without postoperative radiotherapy,and one patient who underwent eye-sparing surgery via sub-brow approach.These four patients all demonstrated a recurrence involving the invasion of extraorbital tissues as metastatic form through surgical bone seams.CONCLUSION:Preserving intact orbital bone tissue is crucial for mitigating direct cross-organ metastasis of lacrimal gland adenoid cystic carcinoma.The findings suggest avoiding the lateral orbitotomy approach with no or limited orbital bone wall invasion.

Rare esophageal carcinoma-primary adenoid cystic carcinoma of the esophagus: A case report

BACKGROUND Esophageal adenoid cystic carcinoma(EACC)is an exceedingly rare malignant tumor of the esophagus,posing significant challenges in the clinic.CASE SUMMARY This report detailed the case of a 72-year-old male whose diagnosis of EACC was confirmed through postoperative histopathological examination.The patient underwent thoracoscopy-assisted radical resection of the esophageal tumor,coupled with lymph node dissection.Pathological findings revealed an adenoid cystic carcinoma infiltrating the entire layer of the muscularis propria,locally extending into the outer membrane of the esophageal fiber,involving the cardia and exhibiting no lymph node metastasis.The patient’s condition was classified as primary EACC,T3N0M0,per the American Joint Committee on Cancer(2017;8th edition).One month after surgery,the patient received postoperative adjuvant radiation therapy.CONCLUSION In addressing the rarity and high potential for biopsy misdiagnosis of EACC,this study delved into its diagnostic methods and treatment.

Ethmoidal Polyposis, Adenoid Hypertrophy and Tympanic Membrane Perforation—A Case Report

Background: Ethmoidal polyposis coexisting with adenoid enlargement and tympanic membrane perforation is a rare condition in otolaryngological practice. Evidences have shown that antrochoanal polyp often times coexists with adenoid enlargement especially in the young which was in sharp contrast to ethmoidal polyposis occurring in the presence of adenoid enlargement. Moreover, the hidden location of nasopharynx housing the adenoids, coupled with the masking effect of bilateral sinonasal polyps, leads to inadvertent misdiagnosis of this complex pathology with subsequent difficulty in providing effective management. Case Presentation: A 24 year-old lady presented with recurrent bilateral nasal obstruction that became persistent associated with nasal discharge and anosmia. She also had recurrent right otorrhoea with associated hearing loss. Diagnostic rigid nasal endoscopy revealed mucoid discharge with bilateral polypoid masses filling both nasal cavities. Otoendoscopic finding revealed a small (about 5%) central tympanic membrane perforation. A clinical assessment of chronic rhinosinusitis with nasal polyposis complicated by chronic suppurative otitis media (CSOM) was made. A non-contrast CT scan of the paranasal sinuses showed isodense lesions in the nasal cavities, all paranasal sinuses and the entire nasopharynx. She had endoscopic sinus surgery and a nasopharyngeal clearance biopsy. The nasal, paranasal and nasopharyngeal masses had histologic confirmation of inflammatory nasal polyps and lymphoid (adenoid) hyperplasia respectively. Her condition improved remarkably with subsequent medical treatment. She was followed up for 8 months and no recurrence was observed. Conclusion: Sinonasal polyposis can coexist with adenoid hypertrophy and middle ear disease as a single pathological condition. Hence, a high index of suspicion and thorough evaluation become necessary for making timely diagnoses and instituting effective management.

Comparison of biological behavior of lacrimal gland adenoid cystic carcinoma with high-grade transformation cells

AIM:To evaluate the differences between human lacrimal gland adenoid cystic carcinoma with high-grade transformation(LACC-HGT)primar y cells cultured by high-grade transformation tissue and non-high-grade transformation(non-HGT)primary cells cultured by non-highgrade transformation tissue in proliferation,metastasis,drug susceptibility,and genes.METHODS:LACC-HGT primary cells were established by tissue block culture,and the 4^(th)to 10^(th)generation primary cells were selected as research objects.The cells were preliminarily identified by immunofluorescent staining.The differences between non-HGT and LACC-HGT primary cells in terms of proliferation,metastasis,and drug susceptibility were compared by cell counting kit-8(CCK-8)assay,wound healing,and drug sensitivity experiments.Differentially expressed genes were screened using mRNA array.Gene expression was analyzed using real-time quantitative polymerase chain reaction(RT-qPCR).RESULTS:LACC-HGT primary cells were successfully cultured by tissue block culture.Immunofluorescence staining results showed that cytokeratin(CK)and CK7 expression levels were positive in LACC-HGT primary cells.CCK-8 results showed that the proliferation ability of LACCHGT cells was significantly higher than that of non-HGT cells.Wound healing experiment showed that the migration ability of LACC-HGT cells was significantly higher than that of non-HGT cells.LACC-HGT cells were also less sensitive to cisplatin and paclitaxel than non-HGT cells.Compared with non-HGT cells,9566 differentially expressed genes were found in LACC-HGT primary cells,of which 5162 were upregulated and 4404 were down-regulated.The expression of N-acetylneuraminate pyruvate lyase(NPL),MARVEL domain containing 3(MARVELD3),syntabulin(SYBU),and allograft inflammatory factor 1(AIF1)was higher in LACCHGT cells than in non-HGT cells,whereas that of periostin(POSTN)was lower.CONCLUSION:LACC-HGT primary cells have faster proliferation,stronger migration ability,and poorer sensitivity to chemotherapy drugs than non-HGT primary cells.The expression of mRNAs in non-HGT and LACC-HGT primary cells are significantly different.These features are speculated to be the reasons why high-grade transformation tissues exhibit higher malignant degree and poorer prognosis than their counterparts.

Preliminary proteomic analysis of human tears in lacrimal adenoid cystic carcinoma and pleomorphic adenoma

·AIM: To detect proteomic differences in tears between adenoid cystic carcinoma(ACC) and pleomorphic adenoma(PA).·METHODS: Tear samples were collected from 4 patients with ACC, 5 with PA, and 4 control cases. Label-free analysis and parallel reaction monitoring(PRM) were used to screen and validate the tear proteome. Gene Ontology(GO) annotation and Kyoto Encyclopedia of Genes and Genomes(KEGG) were conducted for bioinformatics analysis.·RESULTS: In total, 1059 proteins in tear samples were identified by label-free analysis. Between ACC and PA, 415 differentially expressed proteins were detected. Based on the GO annotation, enzyme regulator activity and serinetype endopeptidase inhibitor activity in the molecular function category, blood microparticle and extracellular matrix in the cellular component category, and response to nutrient levels in the biological process category were most predominant. By KEGG pathway annotation, the different proteins between ACC and PA mainly par ticipated in complement and coagulation cascades, amoebiasis, African trypanosomiasis and cholesterol metabolism. Eight proteins with mostly significant differences were verified by PRM, and five proteins with more than 10-fold increases in ACC compared with PA, including integrin β, α-2-macroglobulin, epididymal secretory sperm binding protein Li 78p, RAB5C, and complement C5, were identified.·CONCLUSION: The combined tools of label-free analysis and PRM are very effective and efficient, especially for samples such as tears. Some proteomic dif ferences in tears between ACC and PA are identified and these protein candidates may be specific biomarkers for future exploration.

Rapid response to radiotherapy in unresectable tracheal adenoid cystic carcinoma:A case report

BACKGROUND Adenoid cystic carcinoma(ACC)occurs mainly in the head and neck.Tracheal ACC(TACC)is uncommon.Primary resection is recommended as the main treatment of choice,and radiotherapy is considered for residual disease in the postoperative setting.Definitive radiotherapy is an alternative approach to cure unresectable TACC.As the status of radiosensitivity in TACC is uncertain,the evidence for radiotherapy in unresectable TACC is not well established,especially in terms of the optimal dosage and its response evaluation.Herein,we report a case of locally advanced TACC.CASE SUMMARY A 49-year-old woman was diagnosed with TACC,which included a range of lesions arising in the upper trachea extending caudally 2 cm to 7 cm of the glottis.She was treated with definitive radiotherapy,given the low likelihood of complete resection of the disease.Due to the indolent growth and the propensity for infiltration along the airways,the scheduled radiation dose of 76 Gy in 38 fractions with 6-MV X-ray delivered by intensity-modulated radiotherapy was conducted to the primary tumor volume.After irradiation of 40 Gy,the patient’s dyspnea on exertion was dramatically relieved and bronchoscopy revealed that the previous large polypoid intra-luminal mass was significantly eliminated,with near-complete response.The patient completed two phases of scheduled radiotherapy,and acute reactions to treatment included subjective chest tightness and grade 2 esophagitis,managed medically.After 5 years of treatment,the patient is alive without recurrent disease,and there were no serious late radiation esophagus and lung damage,with only slight dysphagia without perforation and fistula.CONCLUSION Taken together,TACC is uncommon and the treatment of unresectable TACC is challenging.This case indicated that patients with unresectable TACC who rapidly respond to radiation may benefit from primary radical radiotherapy.Radiotherapy may be considered an effective alternative treatment modality.

Adenoid cystic carcinoma of breast: Recent advances

Adenoid cystic carcinoma(ACC) of the breast is a rare special subtype of breast cancer characterized by the presence of a dual cell population of luminal and basaloid cells arranged in specific growth patterns. Most breast cancers with triple-negative, basal-like breast features(i.e., tumors that are devoid of estrogen receptor, progesterone receptor, and human epidermal growth factor receptor 2 expression, and express basal cell markers) are generally high-grade tumors with an aggressive clinical course. Conversely, while ACCs also display a triple-negative, basal-like phenotype, they are usually low-grade and exhibit an indolent clinical behavior. Many discoveries regarding the molecular and genetic features of the ACC, including a specific chromosomal translocation t(6;9) that results in a MYB-NFIB fusion gene, have been made in recent years. This comprehensive review provides our experience with ACC of the breast, as well as an overview of clinical, histopathological, and molecular genetic features.

Nerve Growth Factor and Vascular Endothelial Growth Factor: Retrospective Analysis of 63 Patients with Salivary Adenoid Cystic Carcinoma

Aim To detect the expression of nerve growth factor （NGF） and vascular endothelial growth factor （VEGF） in salivary adenoid cystic carcinoma （SACC） tissues, as well as to determine the correlation between growth factor expression and prognosis in SACC. Methodology Medical records of 63 patients surgically treated for SACC between January 1988 and October 2005 were reviewed. Immunohistochemistry was performed to examine the expression of NGF and VEGF in tumor tissues. Kaplan-Meier analysis and Cox＇s proportional hazard regression model were applied to assess predictors of survival. Results NGF and VEGF were overexpressed in SACC tissues, compared with those in normal salivary tissues （P〈0.05）, and the staining intensity of these two factors was stronger in groups of solid subtype, advanced TNM stage, perineural invasion and recurrence. Patients with high- expression of NGF and VEGF, solid subtype, advanced stage, perineural invasion, recurrence and extended resection alone had worse survival rates （P〈0.05）. Conclusion NGF and VEGF are expressed increasingly in the tissues of SACC cases with invasion and metastasis. NGF expression and VEGF expression are independent prognosis factors for survival.

Association of asymptomatic otitis media with effusion in patients with adenoid hypertrophy

Objective: Clinical symptoms of otitis media with effusion are rarely brought forward to the guardians of young children who the disease is most prevalent in. This often leads to poor scholastic performances and difficult social interactions. The objective of this study was to identify asymptomatic cases of otitis media with effusion present in individuals with adenoid hypertrophy. Material and Methods: In a cross sectional study advocated in Justice K.S.Hegde Hospital, Karnataka India we evaluated one hundred patients above the age of three from August 2016 to December 2017. Candidates who presented with an adenoid nasopharyngeal ratio of more than 0.5 were selected for the study. Individuals who complained of otological symptoms were not considered for the study. Patients cleared of other pathological otological conditions were underwent audiological evaluation with pure tone audiometry and tympanometry for evaluating the middle ear status and hearing loss. Results: The study showed a total of 36% of patients evaluated presented with asymptomatic otitis media with effusion. In candidates who presented with a bilateral B tympanogram, 40% had significant conductive hearing loss of more than 25dB. Conclusion: An objective test such as impedance audiometry in all patients with adenoid hypertrophy would aid in the diagnosis of fluid in the middle ear, so that timely intervention can be done and possible complications be averted.

Endoscopic submucosal dissection in a patient with esophageal adenoid cystic carcinoma

We report the first use of endoscopic submucosal dissection(ESD) for the treatment of a patient with adenoid cystic carcinoma of the esophagus(EACC). An 82-year-old woman visited our hospital for evaluation of an esophageal submucosal tumor. Endoscopic examination showed a submucosal tumor in the middle third of the esophagus. The lesion partially stained with Lugol's solution,and narrow band imaging with magnification showed intrapapillary capillary loops with mild dilatation and a divergence of caliber in the center of the lesion. Endoscopic ultrasound imaging revealed a solid 8 mm × 4.2 mm tumor,primarily involving the second and third layers of the esophagus. A preoperative biopsy was non-diagnostic. ESD was performed to resect the lesion,an 8 mm submucosal tumor. Immunohistologically,tumor cells differentiating into ductal epithelium and myoepithelium were observed,and the tissue type was adenoid cystic carcinoma. There was no evidence of esophageal wall,vertical stump or horizontal margin invasion with p T1 b-SM2 staining(1800 μm from the muscularis mucosa). Further studies are needed to assess the use of ESD for the treatment of patients with EACC.

ANTIMETASTATIC EFFECT OF INTEGRIN IIb/IIIa INHIBITORSON SALIVARY ADENOID CYSTIC CARCINOMA

Objectives: To investigate the relation between metastatic potential of salivary adenoid cystic carcinoma (SACC) and tumor cell-platelet adhesion, and the antimetastatic effect of integrin IIb/IIIa inhibitor on SACC. Methods: Tumor cell-platelet adhesion of highly metastatic SACC-LM, non-highly metastatic SACC-83 and effect of aspirin, arginine-aspartate (RD), magnesium acetylsalicylate on adhesion were studiedin vitro. Antimetastafic effect of aspirin, RD, magnesium acetysalicylate on experimental metastasis of SACC was observedin vivo. Results: The tumor cell-platelet adhesion was stronger in SACC-LM than in SACC-83. Aspirin, RD and magnesium acetylsalicylate could inhibit the adhesion of SACC-LM at the concentration of 1, 5 and 25 μg/ml. RD can inhibit experimental metastasis of SACC. Conclusion: Metastasis of SACC is related to platelet-tumor cell adhesion, RD could inhibit metastasis of SACC.

Expressions of chemokine receptor CXCR4 and its ligand CXCL12 in salivary adenoid cystic carcinoma

Objective: To examine expressions of chemokine receptor CXCR4 and its ligand CXCL12 in primary focus and lymphogenous metastasis of salivary adenoid cystic carcinoma (ACC) with lung metastasis. Methods: Using immunohistochemical hypersensitivity catalyzed signal amplification (CSA), expressions of chemokine receptor CXCR4 and ligand CXCL12 were detected in tissue specimens from 20 cases of primary cancer focus and lymphogenous metastasis of salivary adenoid cystic carcinoma, of which 7 cases were associated with lung metastasis and 3 with lympogenous metastasis. Twenty cases of tongue carcinoma (including 10 cases with lymphogenous metastasis) and 15 cases of mucoepidermoid carcinoma (including 5 cases with lymphogenous metastasis) were used as the malignant control group;and salivary mixed tumor (n=10), tongue leukoceratosis (n=10) and cervical lymph node reactive hyperplasia (n=10) were used as the benign control group. Results: Expression of CXCR4 in the tissues and lymph metastases of oral and maxillofacial salivary ACC, mucoepidermoid carcinoma and tongue carcinoma was significantly higher than that of the benign control group (P<0.05); expression of CXCR4 in the primary focus of ACC was significantly higher than that of the malignant control group; and expression of CXCR4 in the ACC with lung metastasis was 87.1% (6/7), significantly higher than that without lung metastasis(P<0.01). There was evident positive expression of CXCL12 in endotheliocytes of microvessels within cancer and paracancer tissues and significantly high expression of CXCL12 in lymphogenous metastasis(P<0.05). Conclusion: Chemokine receptor CXCR4 and its ligand CXCL12 may be associated with local invasion and lymphogenous metastasis of oral and maxillofacial cancer, especially with lung metastasis of salivary ACC.

Similar therapeutic effects of ^(125I) seed radiotherapy andγ-ray radiotherapy on lacrimal gland adenoid cystic carcinoma

AIM:To evaluate the survival outcomes of patients with lacrimal gland adenoid cystic carcinoma who underwent eye-sparing surgery combined with ^(125I)seed implantation radiotherapy or local externalγ-ray radiotherapy.METHODS:In this retrospective comparative case series,the clinical records of 27 primary and 8 recurrent patients were reviewed.Univariate and multivariate analyses were used to identify risk factors associated with distant metastasis(DM),and the overall survival(OS)after the initial surgery was analyzed.RESULTS:The median follow-up after radiotherapy was 36 mo(range 6-120 mo).At the last follow-up after radiotherapy,26(74.3%)patients had no evidence of disease,7(20%)patients had DM,2(5.9%)patients died of DM,and 1 patient with DM was lost to follow-up.Univariate analyses showed that duration of symptoms,bone destruction,T stage classification,and wide excision surgery were risk factors influencing DM(P<0.05).The 5-year and 10-year OS rates after the initial surgery were 95.8%and 79.9%,respectively.The 5-year DM-free survival and disease-free survival rates after radiotherapy were 66.4%and 52.7%,respectively.CONCLUSION:^(125I)seed radiotherapy and local externalγ-ray radiotherapy may have similar therapeutic effects in preventing DM.Patients with T1/T2 stage disease have a better prognosis than those with T3/T4 stage disease.

Aberrantly expressed GFRα-1/RET in patients with lacrimal adenoid cystic carcinoma is associated with high recurrence risk:a retrospective study of 51 LACC cases

Objective:Because of the poor prognosis of lacrimal adenoid cystic carcinoma(LACC),we aimed to investigate the effects of perineural invasion(PNI)and consequent aberrations in GDNF/GFRα-1/RET protein expression on LACC recurrence.Methods:Clinicopathological data for 51 histologically confirmed patients with LACC enrolled between 2001 and 2017 were retrospectively analyzed.Hematoxylin and eosin staining was applied to assess PNI.Tissue-based immunohistochemistry(IHC)detection of GDNF,GFRα-1,and RET proteins was performed on LACC formalin-fixed,paraffin-embedded specimens.We generated semi-quantitative data of the IHC results and compared them with the clinicopathological data for the 51 patients.Results:Of the 51 patients,19(37.3%)were PNI positive.Recurrence was more common for LACC with than without PNI(73.7%vs.37.5%,P=0.01).GDNF,GFRα-1,and RET proteins were expressed in 62.7%,62.7%,and 54.9%of the 51 patients with LACC,respectively.The expression of all 3 proteins was more common in patients with than without PNI.In agreement with previous findings,PNI-associated GFRα-1 and RET positivity,as detected by IHC,remained significantly associated with recurrence,whereas GDNF expression,as detected by IHC,was not correlated with LACC recurrence.Specifically,patients with concurrent GFRα-1 and RET expression may have a high risk of PNI(89.5%positivity rate)and recurrence(84.2%positivity rate).Conclusions:PNI may contribute to LACC recurrence.The concurrent expression of GFRα-1 and RET proteins,as detected by IHC,may potentially be associated with LACC PNI and recurrence.

RNAi knockdown of C-erbB2 expression inhibits salivary gland adenoid cystic carcinoma SACC-83 cell growthin vitro

Objective：To knockdown the C-erbB2 gene in salivary gland adenoid cystic carcinoma SACC-83 cells using RNA interference,and determine the effect of silencing C-erbB2 on cell proliferation.Methods：C-erbB2-siRNA was transfected into SACC-83 cells.RT-PCR and immunohistochemistry were used to detect C-erbB2 expression in SACC-83 cells.Cell proliferation was measured by the MTT assay and gene knockdown was achieved by RNA interference.Apoptosis was analyzed by flow cytometry.Results：Compared with the control,C-erbB2 mRNA expression was decreased in the C-erbB2-siRNA transfection group,and immunohistochemical analysis indicated that C-erbB2 protein expression was decreased.After C-erbB2-siRNA was transfected for 48 h,absorbance at 570 nm （MTT）was 0.185±0.021 compared with 0.354±0.034,0.299±0.053,and 0.314±0.049 in the blank control,liposome control and negative control siRNA groups,respectively.The differences were statistically significant （P〈0.05）between the C-erbB2-siRNA group and the control groups.Following the C-erbB2 knockdown,the percentage of apoptotic cells was 5.63%compared with 2.04%,2.85%,and 2.98%in the three control groups,respectively.Proliferation of SACC-83 cells was inhibited,and early apoptotic cells were increased.Conclusion： RNA interference can effectively silence C-erbB2 gene expression and inhibit growth of SACC-83 cells,which indicates the potential of targeting this gene as a novel gene therapy approach for the treatment of salivary gland adenoid cystic carcinoma.

Adenoid cystic carcinoma in the maxillary gingiva: a case report and immunohistochemical study

Gingival adenoid cystic carcinoma (ACC) is a rare malignancy. We describe the diagnosis and treatment of a 43 year- old woman who presented with a persistent oral ulcer for approximately 1 year, and subsequent pain in the left posterior maxillary region. Clinical examination revealed an ulcer in the left upper molar gingiva, with swelling in the region from the second premolar to the third molar. X-ray images demonstrated the involvement of the maxillary alveolar bone. The histopathological and immunohistochemical features were diagnostic of ACC. ACC is often presented as a gingival lesion; thus, it may easily be neglected by patients. The identification of this tumor using specific pathological analyses prevents misdiagnosis and enables clinicians to determine the appropriate treatment. In this case, no recurrence or distant metastasis was observed after 2 years of follow-up.

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

Adenoid cystic carcinoma(ACC) of the Bartholin's gland is a rare malignant tumor of vulvar which is characterized by slow growth,local invasion and perineural infiltration.The survival rates for 10 years range from 50% to 100%.The disease free interval for 10 years range from 33% to 38%.Currently,there is no consensus on the treatment of ACC of the Bartholin's gland.Primary surgery includes wide local excision or radical vulvectomy with or without lymph node dissection.Adjuvant radiotherapy and chemotherapy are advocated for the treatment of this cancer.Work is still needed to identify an effective systemic therapy.

Breast adenoid cystic carcinoma arising in microglandular adenosis:A case report and review of literature

BACKGROUND Breast adenoid cystic carcinoma(AdCC)is a rare invasive carcinoma composed of epithelial and myoepithelial cells.Microglandular adenosis(MGA)is a rare benign proliferative lesion consisting of small,uniform,and round glands formed by a single layer of epithelial cells and basement membrane without a myoepithelial cell layer.MGA may progress to atypical MGA and carcinoma arising in MGA.Among various invasive carcinomas from MGA,AdCC has been rarely reported.Here,we report a case of AdCC arising in MGA.CASE SUMMARY A 59-year-old woman was diagnosed with a newly developed density on a routine mammogram.The density was similar to or slightly lower than that of the breast parenchyma.Sonography showed an irregular mass with a slightly higher echo than that of fat.Magnetic resonance imaging showed an irregular mass with a similar T1 signal intensity and a slightly higher T2 signal intensity compared to muscles or the breast parenchyma.The lesion showed heterogeneous internal enhancement with an initially slow and delayed persistent enhancing pattern.Microscopically,the tumor was composed of invasive AdCC,in situ AdCC,and MGA.AdCC is composed of basaloid and ductal epithelial cells forming cribriform or solid sheets,or haphazardly scattered small cribriform or tubular glands.MGA showed small glands with a single epithelial lining and retained lumen.S-100 staining was strongly positive in MGA area.The patient underwent breast-conserving surgery with sentinel lymph node biopsy.CONCLUSION Breast AdCC arising in MGA showed unique imaging findings that was different from usual invasive cancer.