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1.
BMJ Case Rep ; 14(1)2021 Jan 07.
Artigo em Inglês | MEDLINE | ID: mdl-33414116

RESUMO

A 57-year-old Southeast Asian woman with a remote history of adenoid cystic carcinoma (ACC) of the right labium superius oris (upper lip) presented to the hospital with vague epigastric pain. On workup, she was found to have multiple pleural nodules. Histopathology confirmed the diagnosis of metastatic ACC. After 8 months of active surveillance, evidence of disease progression was found and the patient was started on pembrolizumab. Follow-up after starting pembrolizumab showed stable disease with no significant side effects.


Assuntos
Carcinoma Adenoide Cístico/patologia , Neoplasias Labiais/patologia , Neoplasias Pleurais/secundário , Anticorpos Monoclonais Humanizados/uso terapêutico , Antineoplásicos Imunológicos/uso terapêutico , Carcinoma Adenoide Cístico/cirurgia , Diagnóstico Diferencial , Feminino , Humanos , Lábio/patologia , Lábio/cirurgia , Neoplasias Labiais/cirurgia , Pessoa de Meia-Idade , Cavidade Pleural/patologia , Neoplasias Pleurais/diagnóstico , Neoplasias Pleurais/tratamento farmacológico
2.
Cancer Sci ; 112(3): 1184-1195, 2021 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-33377247

RESUMO

Three pathological grading systems advocated by Perzin/Szanto, Spiro, and van Weert are currently used for adenoid cystic carcinoma (AdCC). In these systems, the amount or presence of the solid tumor component in AdCC specimens is an important index. However, the "solid tumor component" has not been well defined. Salivary AdCC cases (N = 195) were collected after a central pathology review. We introduced a novel criterion for solid tumor component, minAmax (minor axis maximum). The largest solid tumor nest in each AdCC case was histologically screened, the maximum oval fitting the solid nest was estimated, and the length of the minor axis of the oval (minAmax) was measured. The prognostic cutoff for the minAmax was determined using training and validation cohorts. All cases were evaluated for the four grading systems, and their prognostic impact and interobserver variability were examined. The cutoff value for the minAmax was set at 0.20 mm. Multivariate prognostic analyses showed the minAmax and van Weert systems to be independent prognostic tools for overall, disease-free, and distant metastasis-free survival while the Perzin/Szanto and Spiro systems were selected for overall survival but not for disease-free or distant metastasis-free survival. The highest hazard ratio for overall survival (11.9) was obtained with the minAmax system. The reproducibility of the minAmax system (kappa coefficient of 0.81) was scored as very good while those of the other three systems were scored as moderate. In conclusion, the minAmax is a simple, objective, and highly reproducible grading system useful for prognostic stratification for salivary AdCC.


Assuntos
Carcinoma Adenoide Cístico/diagnóstico , Neoplasias das Glândulas Salivares/diagnóstico , Glândulas Salivares/patologia , Adulto , Idoso , Idoso de 80 Anos ou mais , Carcinoma Adenoide Cístico/mortalidade , Carcinoma Adenoide Cístico/patologia , Carcinoma Adenoide Cístico/cirurgia , Intervalo Livre de Doença , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Gradação de Tumores/métodos , Prognóstico , Reprodutibilidade dos Testes , Estudos Retrospectivos , Neoplasias das Glândulas Salivares/mortalidade , Neoplasias das Glândulas Salivares/patologia , Neoplasias das Glândulas Salivares/cirurgia , Glândulas Salivares/cirurgia , Adulto Jovem
4.
Rev. senol. patol. mamar. (Ed. impr.) ; 33(3): 108-111, jul.-sept. 2020. ilus
Artigo em Espanhol | IBECS | ID: ibc-197294

RESUMO

El cáncer de mama asociado al embarazo representa una compleja situación clínica por la dificultad de su diagnóstico. La clínica principal del cáncer de mama gestacional es el nódulo palpable, sin embargo, debido a los cambios fisiológicos que se producen en la glándula durante el embarazo o la lactancia, el diagnóstico es especialmente difícil, lo que provoca un retraso en el diagnóstico. El carcinoma escamoso es un subtipo histológico raro, que representa menos del 0,5% de los cánceres de mama. Se trata de un tipo de tumor que suele presentarse en forma de masa quística y cuyo diagnóstico puede confundirse con una entidad benigna. Se presenta el caso de una mujer de 35 años diagnosticada de un carcinoma escamoso de mama tras múltiples ingresos médicos por sospecha de galactoceles y mastitis. El cáncer de mama debe ser el primer diagnóstico diferencial que tener en cuenta ante un nódulo persistente que no mejora tras tratamiento médico y es importante pensar en el carcinoma escamoso en caso de que se trate de una masa quística o absceso mamario


Pregnancy-associated breast cancer represents a complex clinical situation due to the difficulty of diagnosis. The main clinical feature is a palpable nodule. However, due to the physiological changes that occur in the gland during pregnancy or lactation, diagnosis is especially difficult and sometimes delayed. Squamous cell carcinoma is a rare histological subtype, representing less than 0.5% of breast cancers. It usually presents as a cystic mass that can be mistaken for a benign entity. We present the case of a 35-year-old woman diagnosed with squamous cell carcinoma of the breast after multiple medical admissions due to suspicion of galactoceles and mastitis. Breast cancer should be the first differential diagnosis to consider in the presence of a persistent nodule that does not improve after medical treatment and it is important consider squamous cell carcinoma if there is a cystic mass or breast abscess


Assuntos
Humanos , Feminino , Gravidez , Adulto , Complicações Neoplásicas na Gravidez/diagnóstico , Neoplasias da Mama/patologia , Carcinoma de Células Escamosas/patologia , Carcinoma Adenoide Cístico/patologia , Diagnóstico Diferencial , Mastite/patologia
5.
Medicine (Baltimore) ; 99(31): e21531, 2020 Jul 31.
Artigo em Inglês | MEDLINE | ID: mdl-32756201

RESUMO

RATIONALE: Thymic carcinoma with adenoid cystic carcinoma-like features is a special subtype of thymic adenocarcinoma, and the occurrence of this condition is extremely rare. Herein, we report a case of primary thymic carcinoma with adenoid cystic carcinoma-like features in a young man. PATIENT CONCERNS: A 38-year-old man had an incidental finding of space-occupying lesion in the anterior mediastinum during a routine health examination. The patient complained of occasional mild chest tightness during hot weather but had no obvious cough, sputum, chest pain, or fever. Contrast-enhanced computed tomography scan of the chest revealed a space-occupying lesion in the anterior mediastinum, which is likely benign. DIAGNOSIS: The lesion was diagnosed as a primary thymic carcinoma with adenoid cystic carcinoma-like features. INTERVENTION: The patient underwent thoracoscopic resection of left anterior mediastinal mass and enlarged resection of thymectomy and mediastinal fat in our hospital. OUTCOMES: The postoperative course was uneventful. LESSONS: The tissue characteristic of this tumor was extremely similar to that of adenoid cystic carcinoma. A precise pathological examination is extremely important to prevent misdiagnoses of the lesion as adenoid cystic carcinoma or other thymic tumors. Immunohistochemical staining is extremely useful for the pathological and differential diagnoses of this tumor.


Assuntos
Timoma/patologia , Neoplasias do Timo/patologia , Adulto , Carcinoma Adenoide Cístico/patologia , Diagnóstico Diferencial , Humanos , Masculino , Mediastino/patologia , Timoma/diagnóstico , Neoplasias do Timo/diagnóstico
6.
Clin Nucl Med ; 45(9): 716-718, 2020 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-32701807

RESUMO

Adenoid cystic carcinoma is a rare cancer that arises from secretory glands. Metastases are mainly seen in the lungs and bones. Cardiac metastases are extremely rare; only 1 case has previously been described. In this patient, cardiac metastases were suspected based on CT imaging. Tumor imaging by Ga-prostate-specific membrane antigen (PSMA)-11 PET is mainly used in prostate cancer patients but is also of interest in adenoid cystic carcinoma patients because of high PSMA-ligand uptake in salivary glands. Ga-PSMA-11 PET imaging supported the suspicion of cardiac metastases, because the cardiac lesions showed similar tracer uptake compared with other metastases.


Assuntos
Carcinoma Adenoide Cístico/patologia , Ácido Edético/análogos & derivados , Neoplasias Cardíacas/diagnóstico por imagem , Neoplasias Cardíacas/secundário , Oligopeptídeos , Tomografia Computadorizada com Tomografia por Emissão de Pósitrons , Idoso , Humanos , Masculino
7.
Zhonghua Jie He He Hu Xi Za Zhi ; 43(7): 571-576, 2020 Jul 12.
Artigo em Chinês | MEDLINE | ID: mdl-32629556

RESUMO

Objective: To explore the efficacy and safety of the covered airway stent loaded with (125)I seeds for the treatment of tracheal adenoid cystic carcinoma (TACC). Methods: We retrospectively reviewed the clinical data from 8 patients with TACC who had received placement of the covered stent loaded with (125)I seeds between December 2014 and July 2017 in the endoscopic center of the Second Affiliated Hospital of Xiamen Medical College. We compared the difference in the dyspnea index, the diameter of the airway lumen, and the lesion surrounding the airway wall before and after treatment. The complications were also recorded during follow-up. Results: Eight patients underwent successful placement of a total of 11 radioactive stents (2 straight-type stents, 2 L-shape stents, and 7 Y-shape stents, all loaded a total of 243 radioactive particles). Displacement of stents took place within 2 weeks in 2 patients, who were managed with re-stenting and fixation. No further displacement occurred during follow-up. The median time to stent removal was 2.9(interquartile range: 2.3,3.0) months. After stent placement, the dyspnea index was significantly decreased compared with pre-treatment level (mean: 0.1 vs. 3.4, t=8.881, P<0.001). Bronchoscopic re-assessment showed that the residual tumor within the airway was detected in only one patient and that the tumor completely disappeared in the remaining 7 patients. Treatment with stents loaded with radioactive particles yielded smooth and pale airway mucosa with formation of partial scar formation. Chest computed tomography re-assessment demonstrated significantly larger luminal diameter than that before treatment (mean: 13.1 mm vs. 3.3 mm, t=-7.839, P<0.001). The airway wall thickness was notably reduced after treatment (mean: 4.3 mm vs. 14.4 mm, t=7.620, P<0.001). The lesions surrounding the airway wall completely disappeared in 7 patients and decreased for more than 50% in a single patient. The median duration of follow-up was 28.0(interquartile range: 24.8,31.5) months. Recurrence of tumor was documented in a single case within 2 years. Six patients did not experience recurrence within the 2-year follow-up period. No death or severe complications were recorded during follow-up. Conclusion: The (125)I radioactive stent is effective for dilating the stenotic airway and ameliorating the symptoms, and thus might be an effective and safe method for the treatment of TACC. Further studies that explore the efficacy of stents loaded with (125)I particles are needed.


Assuntos
Carcinoma Adenoide Cístico/patologia , Carcinoma Adenoide Cístico/cirurgia , Radioisótopos do Iodo , Stents , Neoplasias da Traqueia/patologia , Neoplasias da Traqueia/cirurgia , Estenose Traqueal/terapia , Obstrução das Vias Respiratórias/etiologia , Carcinoma Adenoide Cístico/complicações , Humanos , Recidiva Local de Neoplasia , Estudos Retrospectivos , Neoplasias da Traqueia/complicações , Resultado do Tratamento
8.
Int J Clin Oncol ; 25(10): 1774-1785, 2020 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-32613404

RESUMO

BACKGROUND: Owing to the low incidence of adenoid cystic carcinoma (AdCC), reliable survival estimates and prognostic factors remained unclarified. METHODS: In this multi-institutional retrospective analysis, we collected 192 AdCC cases, and investigated the impact of clinicopathological factors on clinical outcomes of the patients. All AdCC cases were of salivary gland origin and were surgically treated with curative intent. Diagnoses of AdCC were validated by a central pathology review by expert pathologists. RESULTS: The 5-year overall survival (OS) and disease-free survival (DFS) rates were 92.5 and 50.0%, respectively. Treatment failure occurred in 89 patients (46%) with the distant failures in 65 (34%). Multivariate analysis indicated that pN2 and a pathologically positive surgical margin were independent prognostic factors for both OS and DFS. Histological grade III was an independent prognostic factor for OS. A primary site in the submandibular gland, pT3/4, pN1, and histological grade II were independent prognostic factors for DFS. Postoperative radiation therapy (PORT) improved the locoregional control (LRC) rate. Prophylactic neck dissection was not associated with a better OS or better LRC among patients with cN0. Facial nerve dissection did not improve clinical outcomes in parotid AdCC cases without facial nerve palsy. CONCLUSIONS: A higher TN classification, a pathologically positive surgical margin, and a higher histological grade were associated with a lower OS. PORT improved LRC rates but neck dissection failed to improve clinical outcomes in patients with cN0. As the distant metastasis was frequent, effective systemic therapy is imperative to improve the survival of AdCC patients.


Assuntos
Carcinoma Adenoide Cístico/patologia , Carcinoma Adenoide Cístico/cirurgia , Neoplasias das Glândulas Salivares/patologia , Neoplasias das Glândulas Salivares/cirurgia , Adulto , Idoso , Idoso de 80 Anos ou mais , Carcinoma Adenoide Cístico/mortalidade , Carcinoma Adenoide Cístico/radioterapia , Intervalo Livre de Doença , Feminino , Humanos , Japão , Masculino , Margens de Excisão , Pessoa de Meia-Idade , Análise Multivariada , Esvaziamento Cervical , Recidiva Local de Neoplasia , Neoplasias Parotídeas/mortalidade , Neoplasias Parotídeas/patologia , Neoplasias Parotídeas/radioterapia , Neoplasias Parotídeas/cirurgia , Prognóstico , Estudos Retrospectivos , Neoplasias das Glândulas Salivares/mortalidade , Neoplasias das Glândulas Salivares/radioterapia , Resultado do Tratamento , Adulto Jovem
9.
Rev. senol. patol. mamar. (Ed. impr.) ; 33(2): 68-71, abr.-jun. 2020. ilus
Artigo em Espanhol | IBECS | ID: ibc-197286

RESUMO

INTRODUCCIÓN: El carcinoma medular es diagnosticado en aproximadamente del 1 al 7% de todos los cánceres mamarios y existen pocos casos asociados a una lesión quística. CASO CLÍNICO: Femenino de 34 años con diagnóstico de quiste complejo de mama derecha; histopatológicamente se reporta carcinoma medular en una de las paredes del quiste. CONCLUSIONES: Los tumores malignos de mama en formas quísticas han sido descritos en un 0,3-7%, y pocas veces son carcinomas medulares


INTRODUCTION: Medullary carcinoma is diagnosed in approximately 1% to 7% of all breast cancers and has been rarely associated with a cystic lesion. CASE REPORT: A 34-year-old woman was diagnosed with a complex cyst of the right breast. Histopathological analysis identified medullary carcinoma in one of the walls of the cyst. CONCLUSIONS: Malignant tumours of the breast are rarely found in cystic forms. Only 0.3% to 7% of breast cancer cases have been reported as medullary carcinomas


Assuntos
Humanos , Feminino , Adulto , Neoplasias da Mama/patologia , Carcinoma Medular/patologia , Cisto Mamário/patologia , Carcinoma Adenoide Cístico/patologia , Mamografia/métodos , Ultrassonografia Mamária/métodos , Excisão de Linfonodo/métodos , Mastectomia/métodos , Imuno-Histoquímica/métodos
10.
Oncology ; 98(8): 513-519, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-32408297

RESUMO

Background and summary: Among all vulvar cancers, primary adenoid cystic carcinoma (ACC) of Bartholin's gland is a very rare tumor characterized by a slow growth, a high local aggressiveness, and a remarkable recurrence rate. Due to its rarity, treatment remains a challenge for oncologists and gynecological surgeons. Key message: The present paper reports clinical, radiological, and histological features of ACC of Bartholin's gland and reviews the literature data on the treatment options with a particular focus on the potential role of particle radiation therapy.


Assuntos
Glândulas Vestibulares Maiores/patologia , Carcinoma Adenoide Cístico/patologia , Carcinoma Adenoide Cístico/radioterapia , Neoplasias Vulvares/patologia , Neoplasias Vulvares/radioterapia , Carcinoma Adenoide Cístico/diagnóstico , Carcinoma Adenoide Cístico/tratamento farmacológico , Feminino , Radioterapia com Íons Pesados/métodos , Humanos , Recidiva Local de Neoplasia , Prognóstico , Doenças Raras/diagnóstico , Doenças Raras/patologia , Doenças Raras/radioterapia , Fatores de Risco , Neoplasias Vulvares/diagnóstico , Neoplasias Vulvares/tratamento farmacológico
11.
Biochem Biophys Res Commun ; 526(1): 70-77, 2020 05 21.
Artigo em Inglês | MEDLINE | ID: mdl-32192769

RESUMO

Genetic alterations can drive carcinogenesis. Numerous studies have shown that gene fusion is associated with cancer progression and could provide valuable biomarkers for clinical diagnosis or targets for cancer therapy. Adenoid cystic carcinoma (ACC) is a rare form of adenocarcinoma, characterized by frequent local recurrence and high rates of distant metastasis, ultimately resulting in low survival rates. Owing to the lack of effective therapeutic targets and limited biomarkers for diagnosis, a deeper understanding of the molecular basis of ACC is urgently needed. Here, we show that gene fusion is associated with ACC metastasis. We identified a metastasis suppressor KISS1 fused with a close-by gene, GOLT1A, in highly metastatic ACC cell lines and human specimens. Such fusion blocks KISS1 translation, but not transcription, by introducing 5' upstream open reading frames (uORFs) in the GOLT1A-KISS1 fusion transcript. Deletion of these uORFs rescued KISS1 expression and reduced invasion and migration of metastatic ACC cells. We also detected GOLT1A-KISS1 fusion transcripts in other types of highly metastatic cancer cell lines. Taken together, our results highlight the significance of this novel GOLT1A-KISS1 gene fusion in tumor metastasis and provide a valuable biomarker for clinical diagnosis and future therapeutic targeting of ACC.


Assuntos
Carcinoma Adenoide Cístico/metabolismo , Carcinoma Adenoide Cístico/patologia , Proteínas de Fusão Oncogênica/metabolismo , Sequência de Bases , Carcinoma Adenoide Cístico/genética , Linhagem Celular Tumoral , Regulação Neoplásica da Expressão Gênica , Humanos , Metástase Neoplásica , Proteínas de Fusão Oncogênica/genética , Fases de Leitura Aberta/genética , RNA Mensageiro/genética , RNA Mensageiro/metabolismo , Reprodutibilidade dos Testes , Transcrição Genética
12.
J Cancer Res Clin Oncol ; 146(5): 1343-1350, 2020 May.
Artigo em Inglês | MEDLINE | ID: mdl-32144535

RESUMO

PURPOSE: Adenoid cystic carcinoma (AdCC) is generally slow growing but has highly metastatic potential to distant organs. Several factors and biomarkers are associated with metastasis and treatment outcomes, although further definition is needed. Therefore, this study aimed to evaluate the risk factors for survival and distant metastasis in patients with head and neck AdCC. METHODS: This study included 125 patients with previously untreated AdCC who underwent primary surgery with or without radiotherapy in our tertiary referral centre. Univariate and multivariate Cox proportional hazard regression analyses were used to identify risk factors associated with overall survival (OS), cause-specific survival (CSS), disease-free survival (DFS), and distant metastasis-free survival (DMFS). Factors associated with OS in patients with distant metastasis were separately analysed. RESULTS: During a median follow-up of 9.8 years (range 3.0-22.6 years), 58 patients (46.4%) had distant metastasis and 29 (23.2%) died of disease. Multivariate analyses showed that lymphovascular invasion, lymph node metastasis, and distant metastasis were independent factors of OS and CSS outcomes (all P < 0.05). The T classification and extranodal extension were independent factors of DFS and DMFS outcomes (P < 0.05). After patients presented with distant metastasis, the median survival was 5.8 years. Multivariate analyses showed that extranodal extension and regional recurrence were independent factors of survival after occurrence of distant metastasis (P < 0.05). CONCLUSION: Several clinicopathological factors can predict distant metastasis and survival of patients with AdCC treated with primary surgery. This may promote post-treatment surveillance in patients with AdCC.


Assuntos
Carcinoma Adenoide Cístico/cirurgia , Neoplasias de Cabeça e Pescoço/cirurgia , Adolescente , Adulto , Idoso , Carcinoma Adenoide Cístico/mortalidade , Carcinoma Adenoide Cístico/patologia , Carcinoma Adenoide Cístico/radioterapia , Neoplasias de Cabeça e Pescoço/mortalidade , Neoplasias de Cabeça e Pescoço/patologia , Neoplasias de Cabeça e Pescoço/radioterapia , Humanos , Metástase Linfática , Pessoa de Meia-Idade , Metástase Neoplásica , Recidiva Local de Neoplasia/mortalidade , Recidiva Local de Neoplasia/patologia , Modelos de Riscos Proporcionais , Radioterapia Adjuvante , Fatores de Risco , Taxa de Sobrevida , Adulto Jovem
13.
Cancer Res ; 80(11): 2273-2285, 2020 06 01.
Artigo em Inglês | MEDLINE | ID: mdl-32098781

RESUMO

Lung metastasis is a major factor affecting long-term survival in patients with adenoid cystic carcinoma. Here, we showed that the long noncoding RNA (lncRNA) MRPL23 antisense RNA 1 (MRPL23-AS1) was highly expressed and correlated with lung metastasis and overall survival in patients with salivary adenoid cystic carcinoma (SACC). MRPL23-AS1 positively regulated epithelial-mesenchymal transition by forming an RNA-protein complex with enhancer of zeste homolog 2 (EZH2). MRPL23-AS1 increased the binding of EZH2 and H3K27me3 on the E-cadherin promoter region. Moreover, MRPL23-AS1 levels were higher in exosomes isolated from the blood plasma of patients with SACC, and exosomal MRPL23-AS1 affected pulmonary microvascular endothelial cells in an "exosomecrine" manner. MRPL23-AS1-enriched exosomes increased microvascular permeability and facilitated the metastasis of SACC in vivo. Collectively, these findings highlight a molecular mechanism of lung metastasis in SACC. MRPL23-AS1 may represent a biomarker and target for clinical intervention to control this intractable disease. SIGNIFICANCE: This study identifies a novel metastasis-promoting lncRNA MRPL23-AS1, which mediates the transcriptional silencing of E-cadherin through forming an RNA-protein complex with EZH2.


Assuntos
Carcinoma Adenoide Cístico/genética , Neoplasias Pulmonares/genética , RNA Antissenso/genética , RNA Longo não Codificante/genética , Proteínas de Ligação a RNA/genética , Proteínas Ribossômicas/genética , Neoplasias das Glândulas Salivares/genética , Animais , Antígenos CD/biossíntese , Antígenos CD/genética , Caderinas/biossíntese , Caderinas/genética , Carcinoma Adenoide Cístico/metabolismo , Carcinoma Adenoide Cístico/patologia , Proteína Potenciadora do Homólogo 2 de Zeste/genética , Proteína Potenciadora do Homólogo 2 de Zeste/metabolismo , Exoma , Humanos , Neoplasias Pulmonares/metabolismo , Neoplasias Pulmonares/secundário , Camundongos , Camundongos Endogâmicos NOD , Camundongos SCID , RNA Antissenso/metabolismo , Proteínas de Ligação a RNA/metabolismo , Proteínas Ribossômicas/metabolismo , Neoplasias das Glândulas Salivares/metabolismo , Neoplasias das Glândulas Salivares/patologia , Microambiente Tumoral , Regulação para Cima
14.
Am J Case Rep ; 21: e919781, 2020 Jan 08.
Artigo em Inglês | MEDLINE | ID: mdl-31911575

RESUMO

BACKGROUND Adenoid cystic carcinoma (ACC) is a very rare tumor with a high risk of loco-regional recurrence and potential distant metastases. Until now, only a few cases of renal metastases from ACC have been reported in the literature. CASE REPORT A 64-year-old, Caucasian, non-smoker female, 8 months after being treated by radio-chemotherapy for a squamous cell nasal cavity tumor, presented two renal lesions associated with lung and vertebral metastases. Histology was consisted with a metastasis from an ACC. The histological revision of the primary nasal tumor confirmed a squamous cells carcinoma with an adenoid cystic component that metastasized to the kidney. Renal lesions appeared hypometabolic at the ¹8F-fluorodeoxyglucose (¹8F-FDG) PET scan mimicking a primary renal tumor. The patient underwent a systemic, palliative chemotherapy by a weekly carboplatin/paclitaxel/cetuximab regimen that was well tolerated and allowed a lasting tumor control. CONCLUSIONS The particularity of this case relies on the rarity of renal metastasis from ACC, its difficult diagnosis, and the complexity of its management, as no standard chemotherapy has been validated for metastatic ACC, yet. In our case, a weekly carboplatin/paclitaxel/cetuximab regimen was administered leading to a durable tumor stabilization with an excellent patient's quality of life.


Assuntos
Carcinoma Adenoide Cístico/patologia , Carcinoma de Células Escamosas/patologia , Neoplasias Renais/tratamento farmacológico , Neoplasias Renais/secundário , Neoplasias Nasais/patologia , Carboplatina/uso terapêutico , Carcinoma Adenoide Cístico/terapia , Carcinoma de Células Escamosas/terapia , Cetuximab/uso terapêutico , Quimioterapia Combinada , Feminino , Fluordesoxiglucose F18 , Humanos , Pessoa de Meia-Idade , Cavidade Nasal , Neoplasias Nasais/terapia , Paclitaxel/uso terapêutico , Cuidados Paliativos , Tomografia por Emissão de Pósitrons , Doenças Raras/tratamento farmacológico
15.
Cir Cir ; 88(1): 34-40, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-31967612

RESUMO

Introduction: Adenoid cystic carcinoma (ACC) is a rare tumor, with an annual incidence of 3-4.5 cases/million. Materials and methods: We conducted a descriptive and retrospective study. We included patients diagnosed with ACC of head and neck of a single third-level referral center, from January 2008 to December 2013. Results: A total of 23 patients were included in the study, 10 males (43.5%) and 13 females (56.5%) (F:M ratio 1.3:1). Median age was 52 years. Localization was predominantly observed in the oral cavity (26%) followed by the palate (17.4%). At presentation, 15 patients (65.2%) had clinical Stage II. The most frequent histology was the cribriform type in 10 patients (43.5%). Following resection, positive margins were observed in 15 (65.2%), neural invasion in 8 (34.8%), and lymphovascular invasion in 2 (8.7%) patients. The median time to recurrence in our population was 23 months. Recurrence at 5 years was observed in 15 patients (65%). At 2 years, recurrence was predominantly local in 7 patients (46%); whereas after 2 years, it was predominantly pulmonary 6 patients (40%). Overall, 5 year-survival was 78%. Conclusions: Cystic adenoid carcinoma is characterized by a high recurrence rate. Nevertheless, it has a high overall 5-year survival, which justifies an aggressive treatment.


Assuntos
Carcinoma Adenoide Cístico/patologia , Neoplasias de Cabeça e Pescoço/patologia , Adulto , Carcinoma Adenoide Cístico/terapia , Quimioterapia Adjuvante , Feminino , Neoplasias de Cabeça e Pescoço/terapia , Humanos , Masculino , Neoplasias do Seio Maxilar/patologia , Neoplasias do Seio Maxilar/terapia , Pessoa de Meia-Idade , Neoplasias Bucais/patologia , Neoplasias Bucais/terapia , Terapia Neoadjuvante , Invasividade Neoplásica , Neoplasias Palatinas/patologia , Neoplasias Palatinas/terapia , Radioterapia Adjuvante , Estudos Retrospectivos , Centros de Atenção Terciária , Fatores de Tempo
16.
Oral Dis ; 26(2): 302-312, 2020 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-31793126

RESUMO

OBJECTIVES: This study aimed to explore whether RhoG/Rac1 was involved in migration and invasion of salivary adenoid cystic carcinoma (SACC). MATERIALS AND METHODS: RhoG and Rac1 were evaluated in two SACC cell lines, namely SACC-83 and SACC-LM, with low and high rates of lung metastasis, respectively. Functional changes were evaluated using cell proliferation, transwell, and wound-healing assays, and molecular events were investigated using real-time PCR and Western blot assays. RESULTS: RhoG and Rac1 were highly expressed and more activated in SACC-LM cells than in SACC-83 cells. RhoG overexpression promoted SACC-83 cell migration and invasion through activating Rac1. The knockdown of RhoG or Rac1 partially blocked epiregulin-induced migration and invasion in SACC-83 cells. Epiregulin-induced activation of RhoG/Rac1 in SACC-83 cells was blocked by a Src inhibitor, or an AKT inhibitor or AKT siRNA, or an ERK1/2 inhibitor. Moreover, the epiregulin-induced phosphorylation of AKT and ERK1/2 in SACC-83 cells was blocked by a Src inhibitor, and the epiregulin-induced phosphorylation of ERK1/2 was blocked by an AKT inhibitor or AKT siRNA. Overexpression of activated AKT induced activation of ERK1/2 and RhoG. CONCLUSIONS: RhoG/Rac1 signaling pathway was involved in SACC cell migration and invasion. RhoG/Rac1 at least partially mediated epiregulin/Src/AKT/ERK1/2 signaling to promote SACC cell migration and invasion.


Assuntos
Carcinoma Adenoide Cístico/enzimologia , Carcinoma Adenoide Cístico/patologia , Neoplasias das Glândulas Salivares/enzimologia , Neoplasias das Glândulas Salivares/patologia , Proteínas rac1 de Ligação ao GTP/metabolismo , Proteínas rho de Ligação ao GTP/metabolismo , Linhagem Celular Tumoral , Movimento Celular , Epirregulina/metabolismo , Humanos , Sistema de Sinalização das MAP Quinases/efeitos dos fármacos , Invasividade Neoplásica , Proteínas Proto-Oncogênicas c-akt/antagonistas & inibidores , Proteínas Proto-Oncogênicas c-akt/genética , Proteínas Proto-Oncogênicas c-akt/metabolismo , Transdução de Sinais , Proteínas rac1 de Ligação ao GTP/genética , Quinases da Família src/antagonistas & inibidores , Quinases da Família src/metabolismo
17.
J Thorac Cardiovasc Surg ; 159(6): 2525-2537.e23, 2020 06.
Artigo em Inglês | MEDLINE | ID: mdl-31859073

RESUMO

OBJECTIVE: Synthetic tracheal grafts seeded with autologous bone marrow-mononuclear cells (BM-MNCs) have been described as becoming living and functional grafts representing a promising option for tracheal replacement for pathologies unamenable by segmental resection or autologous repair. This study aimed to present the first long-term follow-up of these procedures in humans. METHODS: We retrospectively analyzed 3 patients who received synthetic tracheal grafts seeded with BM-MNCs implanted. RESULTS: Patient 1 was a 37-year-old man with mucoepidermoid carcinoma, the first-ever human to receive a synthetic tracheal graft seeded with BM-MNCs. Patient 2 was a 30-year-old man with adenoid cystic carcinoma, and patient 3 was a 22-year-old woman with an iatrogenic tracheal injury. All patients developed graft-related complications necessitating multiple surgical reinterventions. Patient 1 was hospitalized for 8 months before dying from respiratory failure secondary to graft dehiscence 32 months after implantation. Patient 2 died 3.5 months after implantation from undisclosed causes. Patient 3 received a second synthetic tracheal graft after 11 months and an allogeneic trachea and lung transplantation 45 months after the primary implantation. Patient 3 underwent 191 surgical interventions after the primary implantation and spent 55 months in the intensive care unit before dying from airway bleeding. All patients' bronchoscopic, histologic, and radiologic investigations demonstrated graft-associated complications, including anastomotic fistulae and obstructive granulation tissue, without graft vascularization, mucosal lining, or integration into adjacent tissues. CONCLUSIONS: Synthetic tracheal grafts seeded with BM-MNCs do not become living functional tracheal grafts and lead to debilitating complications and death.


Assuntos
Células da Medula Óssea/fisiologia , Transplante de Medula Óssea , Carcinoma Adenoide Cístico/cirurgia , Carcinoma Mucoepidermoide/cirurgia , Doença Iatrogênica , Traumatismos Torácicos/cirurgia , Engenharia Tecidual , Tecidos Suporte , Traqueia/transplante , Neoplasias da Traqueia/cirurgia , Adulto , Carcinoma Adenoide Cístico/diagnóstico por imagem , Carcinoma Adenoide Cístico/patologia , Carcinoma Mucoepidermoide/diagnóstico por imagem , Carcinoma Mucoepidermoide/patologia , Células Cultivadas , Evolução Fatal , Feminino , Humanos , Masculino , Estudos Retrospectivos , Traumatismos Torácicos/diagnóstico por imagem , Traumatismos Torácicos/patologia , Traqueia/diagnóstico por imagem , Traqueia/lesões , Traqueia/patologia , Neoplasias da Traqueia/diagnóstico por imagem , Neoplasias da Traqueia/patologia , Transplante Autólogo , Resultado do Tratamento , Adulto Jovem
18.
Am J Surg Pathol ; 44(2): 214-223, 2020 02.
Artigo em Inglês | MEDLINE | ID: mdl-31567278

RESUMO

Mammary adenoid cystic carcinoma (ACC) is a rare subtype of breast cancer with a favorable prognosis. Here we report on predictors of outcome based on a detailed morphologic review and analysis of 108 mammary ACC. Sixty-four tumors (59.2%) were pure conventional ACC, 23 (21.3%) were pure basaloid ACC. Follow-up was available for 87 patients (median: 51 mo). Eighteen patients (20.7%) developed recurrence: 7 (8%) had local recurrence and 14 (16%) had distant metastasis. Two patients died of disease, 1 died of an unrelated cause, 14 were alive with disease (including 8 in palliative care), and 70 (80.5%) were alive with no evidence of disease. Of 90 patients with known lymph node (LN) status 9 (10%) had nodal involvement (all with basaloid ACC). Distant metastases in patients with predominantly basaloid ACC compared with pure conventional ACC were more common (40% vs. 7.7%) and occurred earlier (22 vs. 84 mo). The following factors were found to be predictive of recurrence-free survival: positive margin, Nottingham grade, neovascularization, basaloid component, perineural invasion, lymphovascular invasion, >30% solid growth, necrosis and LN involvement; the first 3 remained statistically significant on multivariate analysis. Factors predictive of distant disease-free survival were neovascularization, Nottingham grade, lymphovascular invasion, solid component >50%, LN involvement, basaloid component >50%, tumor necrosis, perineural invasion, and final margin. Only neovascularization remained statistically significant on multivariate analysis. Basaloid ACC is an aggressive variant of mammary ACC with more frequent nodal involvement and higher incidence of distant spread. LN staging should be performed for all mammary basaloid ACC.


Assuntos
Neoplasias da Mama/patologia , Carcinoma Adenoide Cístico/patologia , Adulto , Idoso , Idoso de 80 Anos ou mais , Neoplasias da Mama/diagnóstico , Neoplasias da Mama/mortalidade , Neoplasias da Mama/terapia , Carcinoma Adenoide Cístico/diagnóstico , Carcinoma Adenoide Cístico/mortalidade , Carcinoma Adenoide Cístico/terapia , Terapia Combinada , Feminino , Seguimentos , Humanos , Metástase Linfática , Masculino , Pessoa de Meia-Idade , Gradação de Tumores , Recidiva Local de Neoplasia/diagnóstico , Recidiva Local de Neoplasia/epidemiologia , Recidiva Local de Neoplasia/patologia , Estadiamento de Neoplasias , Prognóstico , Análise de Sobrevida
19.
Int J Radiat Oncol Biol Phys ; 106(1): 167-173, 2020 01 01.
Artigo em Inglês | MEDLINE | ID: mdl-31586664

RESUMO

PURPOSE: The adenoid cystic carcinoma (ACC), Erbitux, and Particle Therapy (ACCEPT) phase 1/2 trial (NCT01192087) evaluated a combined-modality approach (concurrent cetuximab and intensity modulated radiation therapy with carbon ion boost) for newly diagnosed nonmetastatic head and neck ACC. METHODS AND MATERIALS: Twenty-three patients with ACC were enrolled between June 2012 and June 2017 after initial diagnosis or postoperatively. All received a 400 mg/m2 cetuximab loading dose a week before radiation therapy, followed by weekly 250 mg/m2 doses starting on the first day of radiation therapy. The carbon ion radiation therapy boost was 24 Gy (relative biological effectiveness) in 8 daily fractions, followed by intensity modulated radiation therapy (54 Gy). The primary endpoint was safety and feasibility (defined based on Common Terminology Criteria for Adverse Events grade ≥3 events). Secondary endpoints included local and distant relapse, disease-free survival, and overall survival. RESULTS: Disease was most commonly in the paranasal sinuses (30%), palate (17%), and nasopharynx (17%). Nine (39%) patients underwent surgery (R1: 22%, R2: 78%). Median follow-up was 38.5 months. No patients experienced grade 4 to 5 events. Rates of grade 3 rash and radiation dermatitis were 17% and 22%, respectively. Grade 2 and 3 mucositis and dysgeusia occurred in 43% and 48% and in 9% and 0%, respectively. Grade 2 to 3 dysphagia and xerostomia were present in 43% and 4% and in 26% and 0%, respectively. At last follow-up, 5 (22%) patients experienced in-field relapse and 6 (26%) developed distant metastases. The 3-year disease-free survival was 67%, and median overall survival was 54 months. CONCLUSIONS: Outcomes of this trial were satisfactory. Although the trial did not meet the predefined criteria of feasibility owing to the comparatively high rates of grade 3 dermatitis, numbers are comparable to existing data on cetuximab + radiation therapy.


Assuntos
Antineoplásicos Imunológicos/uso terapêutico , Carcinoma Adenoide Cístico/terapia , Cetuximab/uso terapêutico , Quimiorradioterapia/métodos , Neoplasias de Cabeça e Pescoço/terapia , Radioterapia com Íons Pesados , Radioterapia de Intensidade Modulada , Antineoplásicos Imunológicos/administração & dosagem , Carcinoma Adenoide Cístico/patologia , Cetuximab/administração & dosagem , Quimiorradioterapia/efeitos adversos , Terapia Combinada/métodos , Transtornos de Deglutição/etiologia , Intervalo Livre de Doença , Fracionamento da Dose de Radiação , Esquema de Medicação , Estudos de Viabilidade , Feminino , Neoplasias de Cabeça e Pescoço/patologia , Radioterapia com Íons Pesados/efeitos adversos , Humanos , Masculino , Pessoa de Meia-Idade , Radiodermatite/etiologia , Radioterapia de Intensidade Modulada/efeitos adversos , Eficiência Biológica Relativa , Xerostomia/etiologia
20.
Neurochirurgie ; 66(1): 45-49, 2020 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-31634508

RESUMO

BACKGROUND: Adenoid cystic carcinoma (ACC), or cylindroma, is a rare malignancy believed to arise from epithelial cells of salivary glands. It is a slow-growing but aggressive tumor with a propensity for perineural invasion. Metastases are common to the lung, but rare to the spine. The natural history of ACC spine metastases is unknown and progression is unpredictable. METHODS: (1) A case report was described for a patient diagnosed with spine ACC metastasis of the T6/T7 vertebrae. (2) A literature search was conducted on Medline via PubMed and the Cochrane databases according to the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines to identify articles from 1973 to March 2019 using following keywords: adenoid cystic carcinoma, cylindroma, metastasis, salivary glands. RESULTS/CASE REPORT: We report an unusual case of spine ACC metastasis of the T6/T7 vertebrae in a patient suffering from progressive radiculopathy and thoracic pain. Based on these findings, the patient underwent T6/T7 laminectomy. The diagnosis was confirmed by the pathology results. The primary parotid tumor had been resected 35 years before spine metastasis. A review of literature encompassing 40 years identified 16 patients treated for ACC spine metastases. Mean time to spine metastases was 4.1 years after primary treatment of ACC. Spine metastases were isolated, without local recurrence at the neck. CONCLUSION: This case highlights the critical importance of long follow up of ACC patients, due to late spine metastases. Diagnosis spinal metastasis of salivary carcinoma should be considered, to guide management, especially in clinical follow-up.


Assuntos
Carcinoma Adenoide Cístico/secundário , Neoplasias da Coluna Vertebral/secundário , Idoso , Carcinoma Adenoide Cístico/patologia , Carcinoma Adenoide Cístico/cirurgia , Feminino , Humanos , Síndromes Neoplásicas Hereditárias , Procedimentos Neurocirúrgicos , Neoplasias Parotídeas/patologia , Neoplasias Parotídeas/cirurgia , Neoplasias Cutâneas , Neoplasias da Coluna Vertebral/patologia , Neoplasias da Coluna Vertebral/cirurgia
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