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1.
BMJ Case Rep ; 14(2)2021 Feb 04.
Artigo em Inglês | MEDLINE | ID: mdl-33541998

RESUMO

Rosai-Dorfman disease is a rare benign histiocytic proliferative disease of unknown cause that, in exceptional cases, presents with lesions confined to the skin. Clinically variable types of lesions such as papules, nodules and plaques have been reported. We present a case of a 27-year-old woman with a 1-year history of erythematous papular and nodular lesions on the malar and right axillary regions, previously misdiagnosed as acne. She reported no fever, malaise or weight loss, while physical examination and laboratory workup were normal. Bacteriological and mycobacteriological cultures were negative. Histopathological findings showed dense infiltration of inflammatory cells involving the entire dermis, consisting of large macrophages with emperipolesis, S100 and CD68 positive, neutrophils, eosinophils, lymphocytes and plasma cells. The patient was treated with oral prednisolone without improvement. Dapsone was subsequently initiated with favourable clinical response. The present article aimed to emphasise the clinical and histological differential diagnosis and share the treatment experience.


Assuntos
Anti-Infecciosos/uso terapêutico , Dapsona/uso terapêutico , Glucocorticoides/uso terapêutico , Histiocitose Sinusal , Prednisolona/uso terapêutico , Doenças Raras , Pele/patologia , Adulto , Erros de Diagnóstico , Emperipolese , Eosinófilos/patologia , Feminino , Histiocitose Sinusal/diagnóstico , Histiocitose Sinusal/tratamento farmacológico , Humanos , Proteínas S100
2.
Int J Surg Pathol ; 29(4): 427-432, 2021 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-32909465

RESUMO

Biphasic squamoid alveolar renal cell carcinoma (BSARCC) is a recently described kidney cancer entity with <60 published cases so far. Some unique features of BSARCC include distinct squamoid and alveolar morphology forming glomeruloid configurations and 2 types of tumor cells with different immunoprofiles. Although the mechanism is unknown, neutrophils and cellular materials engulfed by larger tumor cells (described as "emperipolesis") are observed in all reported cases including the current one. In this article, we report a case of a 70-year-old man who presented with an incidental renal mass during workup for immunoglobulin M monoclonal gammopathy of unknown significance and cold agglutinin autoimmune hemolytic anemia. A detailed pathologic evaluation and immunohistochemical studies revealed BSARCC colliding with atypical CD5+ monoclonal B-cells.


Assuntos
Linfócitos B/patologia , Carcinoma de Células Renais/diagnóstico , Neoplasias Renais/diagnóstico , Rim/patologia , Idoso , Linfócitos B/metabolismo , Biomarcadores Tumorais/análise , Biomarcadores Tumorais/metabolismo , Antígenos CD5/análise , Antígenos CD5/metabolismo , Carcinoma de Células Renais/cirurgia , Diagnóstico Diferencial , Emperipolese , Humanos , Achados Incidentais , Rim/citologia , Rim/diagnóstico por imagem , Rim/cirurgia , Neoplasias Renais/patologia , Neoplasias Renais/cirurgia , Masculino , Nefrectomia , Tomografia Computadorizada por Raios X
5.
Diagn Cytopathol ; 48(12): E22-E26, 2020 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-32621636

RESUMO

Emperipolesis is a physiologic or pathologic phenomenon characterized by the presence of intact viable cells within the cytoplasm of another cell. It has been described in normal tissues and in a variety of inflammatory and neoplastic lesions such as Rosai-Dorfman disease, tumors, hematopoietic disorders and rarely lymphomas. Emperipolesis by mesothelial cells is rare. Few cases of mesothelial emperipolesis of neoplastic lymphocytes in pleural effusions involved by lymphomas have been reported in the literature. Its etiopathogenesis and significance are controversial and speculative. We report a case of a 36-year-old man who presented with cough, chest pain, breathing difficulty, pericardial, and bilateral pleural effusions secondary to mediastinal T-lymphoblastic lymphoma. Pleural fluid cytology slides and cell block sections showed numerous single dispersed neoplastic lymphoblasts with occasional giant multinucleated mesothelial cells with emperipolesis of lymphocytes. The background showed scattered and clumped apoptotic karyorrhexis debris and reactive mesothelial cells. Cell block immunohistochemistry showed CD3, CD5, CD7, CD10, CD99, and TdT positive lymphocytes, consistent with involvement by T-lymphoblastic lymphoma. The giant cells were positive for cytokeratin, calretinin and WT1 confirming their mesothelial origin. Lymphoid effusions with emperipolesis may raise a potential diagnostic pitfall because they may morphologically be confused with other inflammatory and neoplastic lesions. This cell-in-cell phenomenon can be a helpful clue in the differential diagnosis of lymphocyte-rich effusions since it has been described in association with lymphomas. It might shed some light on the lymphocyte-mesothelial interaction and the potential phagocytic antigen-presenting properties of mesothelial cells under certain circumstances.


Assuntos
Emperipolese/fisiologia , Epitélio/patologia , Linfócitos/patologia , Neoplasias Mesoteliais/patologia , Derrame Pleural/patologia , Leucemia-Linfoma Linfoblástico de Células Precursoras/patologia , Adulto , Biomarcadores Tumorais/metabolismo , Epitélio/metabolismo , Humanos , Linfócitos/metabolismo , Masculino , Neoplasias Mesoteliais/metabolismo , Derrame Pleural/metabolismo , Leucemia-Linfoma Linfoblástico de Células Precursoras/metabolismo
6.
Front Immunol ; 11: 650, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-32528462

RESUMO

The liver is our largest internal organ and it plays major roles in drug detoxification and immunity, where the ingestion of extracellular material through phagocytosis is a critical pathway. Phagocytosis is the deliberate endocytosis of large particles, microbes, dead cells or cell debris and can lead to cell-in-cell structures. Various types of cell endocytosis have been recently described for hepatic epithelia (hepatocytes), which are non-professional phagocytes. Given that up to 80% of the liver comprises hepatocytes, the biological impact of cell-in-cell structures in the liver can have profound effects in liver regeneration, inflammation and cancer. This review brings together the latest reports on four types of endocytosis in the liver -efferocytosis, entosis, emperipolesis and enclysis, with a focus on hepatocyte biology.


Assuntos
Compartimento Celular/fisiologia , Emperipolese/fisiologia , Endocitose/fisiologia , Entose/fisiologia , Hepatócitos/fisiologia , Fígado/citologia , Animais , Humanos , Imunidade , Inativação Metabólica , Fígado/metabolismo , Regeneração Hepática , Fagocitose
7.
Reumatol. clín. (Barc.) ; 16(2,pt.2): 174-176, mar.-abr. 2020. ilus
Artigo em Inglês | IBECS | ID: ibc-194343

RESUMO

Rosai-Dorfman disease (RDD) is uncommon in daily practice, but needs to be ruled out in rheumatologic conditions to elucidate a wide differential diagnosis. Beside its typical presentation, soft tissue masses can be easily seen in our Rheumatology clinics. Ultrasonography widely extended in our specialty, could also play a role in the diagnosis, to end up with the histological confirmation of the disease


La enfermedad Rosai-Dorfman (ERD) es infrecuente en nuestra práctica diaria, aunque se debe considerar en el diagnóstico diferencial de diferentes procesos en reumatología. La presentación en forma de masa de partes blandas incluye el estudio radiológico y la ecografía de partes blandas muy extendida en nuestra especialidad y, finalmente, la confirmación histológica


Assuntos
Humanos , Feminino , Pessoa de Meia-Idade , Histiocitose Sinusal/diagnóstico por imagem , Histiocitose Sinusal/patologia , Tela Subcutânea/patologia , Antebraço/diagnóstico por imagem , Diagnóstico Diferencial , Antebraço/patologia , Biópsia Guiada por Imagem , Emperipolese/imunologia
8.
Int J Pediatr Otorhinolaryngol ; 131: 109851, 2020 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-31901484

RESUMO

OBJECTIVES: To systematically evaluate the clinico-diagnostic profile and management outcomes of otorhinolaryngologic manifestations of Extranodal Rosai-Dorfman Disease (ENRDD) in the pediatric population. METHODS: The search terms Rosai Dorfman Disease and Sinus Histiocytosis were used to query PubMed, Ovid/Medline, and Scopus databases from inception through September 30, 2018. Studies were systematically reviewed in accordance to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) protocol. All reports of ENRDD involving at least one otorhinolaryngologic subsite in children less than 18 years were eligible for inclusion. RESULTS: A total of 31 studies met inclusion criteria resulting in identification of 31 ENRDD cases with a mean age of 11.5 years. Of the 31 patients, 23 were male (74.2%) and 7 were female (22.6%). Extranodal lesions were limited to otorhinolaryngologic subsites in 24 patients (77.4%), while 7 patients (22.5%) were found to have extranodal lesions simultaneously involving otorhinolaryngologic and nonotorhinolaryngologic sites. The nasal vault was the most common otorhinolaryngologic site involved (n = 13, 41.9%), followed by the paranasal sinuses (n = 10, 32.3%). The most common non-otorhinolaryngologic site that was concurrently involved was the orbit (n = 4, 57.1%). Concurrent cervical lymphadenopathy was present in 19 patients (61.2%). While not documented for 2 cases, emperipolesis on histopathology was confirmed in 29 patients (93.5%). Single therapy with surgical excision was the most common modality of treatment (n = 15, 53.6%) and yielded highest remission rates (80%). In 11 instances (34.36%), ENRDD was misdiagnosed. CONCLUSION: Pediatric ENRDD is a rare disease entity that maintains a high misdiagnosis potential. The most common otorhinolaryngologic location for extranodal manifestation is the sinonasal compartment. Surgical excision remains the most common treatment modality yielding lowest persistence and/or recurrence rates.


Assuntos
Histiocitose Sinusal/complicações , Doenças Nasais/etiologia , Doenças Orbitárias/etiologia , Doenças dos Seios Paranasais/etiologia , Criança , Erros de Diagnóstico , Emperipolese , Cabeça , Histiocitose Sinusal/diagnóstico , Histiocitose Sinusal/patologia , Histiocitose Sinusal/cirurgia , Humanos , Linfadenopatia/etiologia , Cavidade Nasal , Pescoço
9.
Ann Diagn Pathol ; 45: 151446, 2020 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-31978810

RESUMO

Primary cutaneous Rosai-Dorfman disease is a rare form of Rosai-Dorfman disease limited to the skin. The diagnosis of primary cutaneous disease is based on a combination of clinical presentation, histopathology, and the detection of S100+, CD68+, and CD1a- histiocytic immunophenotyping. However, the diagnosis of primary cutaneous disease is often difficult and significantly delayed due to the non-specific nature of its histologic and clinical features. In this review, we describe four cases in order to familiarize pathologists and dermatopathologists with the clinicopathologic correlation of primary cutaneous Rosai-Dorfman disease and to help facilitate early diagnosis. In addition, we discuss the proposed pathophysiology and molecular etiology of this tumor, and its relationship with IgG4 sclerosing disease.


Assuntos
Histiocitose Sinusal/metabolismo , Histiocitose Sinusal/patologia , Imunofenotipagem/métodos , Dermatopatias/patologia , Administração Tópica , Adulto , Idoso , Antígenos CD/metabolismo , Antígenos CD1/metabolismo , Antígenos de Diferenciação Mielomonocítica/metabolismo , Biópsia , Diagnóstico Diferencial , Emperipolese , Feminino , Histiócitos/patologia , Histiocitose Sinusal/fisiopatologia , Histiocitose Sinusal/terapia , Humanos , Injeções Intralesionais , Linfadenopatia/patologia , Masculino , Pessoa de Meia-Idade , Proteínas S100/metabolismo , Esteroides/administração & dosagem , Esteroides/uso terapêutico
11.
Acta Cytol ; 64(3): 274-278, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-31527381

RESUMO

Emperipolesis is a biological process defined by the presence of an intact cell within the cytoplasm of another cell. In pleural fluid cytology, the phenomenon is very rare but has already been described in B-cell lymphoma. Here, we report the first case of a T-cell lymphoma diagnosed on a pleural fluid exhibiting extensive emperipolesis of tumor cells by mesothelial cells. Additionally, in order to evaluate the prevalence of emperipolesis in lymphocyte rich pleural effusion cytology, we reviewed cases from our archive, including cases with lymphoma and cases with reactive T-lymphocyte rich effusion.


Assuntos
Emperipolese , Células Epiteliais/patologia , Neoplasias do Mediastino/patologia , Derrame Pleural Maligno/patologia , Leucemia-Linfoma Linfoblástico de Células T Precursoras/patologia , Citodiagnóstico , Humanos , Linfócitos/patologia , Masculino , Pessoa de Meia-Idade
12.
Int J Gynecol Pathol ; 39(5): 443-446, 2020 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-31274698

RESUMO

Sinus histiocytosis with massive lymphadenopathy, also known as Rosai-Dorfman disease (RDD), is a self-limited histiocytic disorder of unclear etiology which most commonly presents with cervical lymphadenopathy. Purely extranodal presentation of RDD is uncommon, and isolated intralymphatic/intravascular confinement of this entity has not previously been described. We report a 16-yr-old female who presented with vaginal swelling and mass-like enlargement of the right labia. The mass had been present for nearly a year without pain or tenderness. Clinically, the lesion was thought to be a Bartholin gland cyst. Following surgical resection, histologic examination demonstrated a hypocellular myxedematous stroma with a mixture of ectatic thin and thick-walled vessels within which there were numerous collections of histiocytes, lymphocytes, and plasma cells. The histopathologic differential diagnosis included localized vulvar lymphedema, a specialized genital tract neoplasm, and childhood asymmetric labium majus enlargement. The histiocytes showed occasional plasma cells and lymphocytes within their cytoplasm, consistent with emperipolesis. Immunohistochemical studies showed that the histiocytes expressed CD163 and S100, while ERG and D2-40 highlighted their intralymphatic confinement, ultimately leading to the diagnosis of intralymphatic RDD. Intralymphatic RDD may present as vulvar lymphedema and can potentially mimic other myxedematous neoplasms of the vulvovaginal region.


Assuntos
Histiocitose Sinusal/diagnóstico , Linfedema/diagnóstico , Neoplasias Vulvares/diagnóstico , Adolescente , Diagnóstico Diferencial , Emperipolese , Feminino , Histiocitose Sinusal/complicações , Histiocitose Sinusal/patologia , Humanos , Imuno-Histoquímica , Linfedema/complicações , Linfedema/patologia , Linfócitos/patologia , Plasmócitos/patologia , Vagina/patologia , Neoplasias Vulvares/complicações , Neoplasias Vulvares/patologia
14.
Platelets ; 31(6): 700-706, 2020 Aug 17.
Artigo em Inglês | MEDLINE | ID: mdl-31752579

RESUMO

Histology of bone marrow routinely identifies megakaryocytes that enclose neutrophils and other hematopoietic cells, a phenomenon termed emperipolesis. Preserved across mammalian species and enhanced with systemic inflammation and platelet demand, the nature and significance of emperipolesis remain largely unexplored. Recent advances demonstrate that emperipolesis is in fact a distinct form of cell-in-cell interaction. Following integrin-mediated attachment, megakaryocytes and neutrophils both actively drive entry via cytoskeletal rearrangement. Neutrophils enter a vacuole termed the emperisome which then releases them directly into the megakaryocyte cytoplasm. From this surprising location, neutrophils fuse with the demarcation membrane system to pass membrane to circulating platelets, enhancing the efficiency of thrombocytogenesis. Neutrophils then egress intact, carrying megakaryocyte membrane and potentially other cell components along with them. In this review, we summarize what is known about this intriguing cell-in-cell interaction and discuss potential roles for emperipolesis in megakaryocyte, platelet and neutrophil biology.


Assuntos
Emperipolese/genética , Megacariócitos/patologia , Neutrófilos/metabolismo , Humanos
15.
J Cell Mol Med ; 24(2): 1268-1275, 2020 01.
Artigo em Inglês | MEDLINE | ID: mdl-31851780

RESUMO

Primary biliary cholangitis (PBC) is an autoimmune disease characterized by chronic destruction of the bile ducts. A major unanswered question regarding the pathogenesis of PBC is the precise mechanisms of small bile duct injury. Emperipolesis is one of cell-in-cell structures that is a potential histological hallmark associated with chronic hepatitis B. This study aimed to clarify the pathogenesis and characteristics of emperipolesis in PBC liver injury. Sixty-six PBC patients, diagnosed by liver biopsy combined with laboratory test, were divided into early-stage PBC (stages I and II, n = 39) and late-stage PBC (stages III and IV, n = 27). Emperipolesis was measured in liver sections stained with haematoxylin-eosin. The expressions of CK19, CD3, CD4, CD8, CD20, Ki67 and apoptosis of BECs were evaluated by immunohistochemistry or immunofluorescence double labelling. Emperipolesis was observed in 62.1% of patients with PBC, and BECs were predominantly host cells. The number of infiltrating CD3+ and CD8+ T cells correlated with the advancement of emperipolesis (R2  = 0.318, P < .001; R2  = 0.060, P < .05). The cell numbers of TUNEL-positive BECs and double staining for CK19 and Ki67 showed a significant positive correlation with emperipolesis degree (R2  = 0.236, P < .001; R2  = 0.267, P < .001). We conclude that emperipolesis mediated by CD8+ T cells appears to be relevant to apoptosis of BEC and thus may aggravate the further injury of interlobular bile ducts.


Assuntos
Apoptose , Ductos Biliares/patologia , Linfócitos T CD8-Positivos/imunologia , Emperipolese , Células Epiteliais/patologia , Cirrose Hepática Biliar/fisiopatologia , Ductos Biliares/imunologia , Ductos Biliares/lesões , Estudos de Casos e Controles , Proliferação de Células , Células Epiteliais/imunologia , Feminino , Humanos , Masculino , Pessoa de Meia-Idade
17.
Am J Surg Pathol ; 43(12): 1653-1660, 2019 12.
Artigo em Inglês | MEDLINE | ID: mdl-31436555

RESUMO

Rosai-Dorfman disease (RDD) is an uncommon disorder, characterized by an atypical expansion of histiocytes which classically shows emperipolesis and immunoreactivity with S-100 protein. RDD affects the lymph nodes as well as extranodal sites; however, RDD of the breast is exceptionally rare. Herein, we describe the histopathologic features of 22 cases of RDD occurring in the breast, with an emphasis on the differential diagnosis. All cases were notable for an exuberant lymphocytic infiltrate with and without germinal center formation, and the majority (19/22) showed numerous plasma cells: 5 to 132/high-power field (HPF). IgG and IgG4 immunohistochemical stains were available for 13 cases; in no instance were criteria for IgG4-related sclerosing disease met, though in a single case the IgG4/IgG ratio was increased to 25%. Sclerosis was present in the majority of cases (18/22), and was frequently prominent. RDD cells showing emperipolesis were present in all cases (22/22), and ranged from rare (<1/50 HPF) to numerous (>50/50 HPF). Two of the cases in our series were initially misdiagnosed as inflammatory myofibroblastic tumor and plasma cell mastitis with granulomatous inflammation. As emperipolesis can be indistinct, the presence of stromal fibrosis and a prominent lymphoplasmacytic inflammatory infiltrate should prompt a careful search for the characteristic histiocytes, which can be aided by the use of S-100 immunohistochemistry.


Assuntos
Doenças Mamárias/imunologia , Mama/imunologia , Histiocitose Sinusal/imunologia , Imunoglobulina G/análise , Neoplasias Inflamatórias Mamárias/imunologia , Mastite/imunologia , Plasmócitos/imunologia , Adolescente , Adulto , Idoso , Mama/química , Mama/patologia , Doenças Mamárias/metabolismo , Doenças Mamárias/patologia , Diagnóstico Diferencial , Emperipolese , Feminino , Fibrose , Histiocitose Sinusal/metabolismo , Histiocitose Sinusal/patologia , Humanos , Neoplasias Inflamatórias Mamárias/química , Neoplasias Inflamatórias Mamárias/patologia , Mastite/metabolismo , Mastite/patologia , Pessoa de Meia-Idade , Plasmócitos/química , Plasmócitos/patologia , Prognóstico , Proteínas S100/análise , Estados Unidos , Adulto Jovem
18.
J Am Soc Cytopathol ; 8(4): 190-205, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31272602

RESUMO

INTRODUCTION: Rosai-Dorfman disease (RDD) is a rare usually self-limited non-Langerhans cell histiocytosis of unknown etiology. Nodal and extranodal RDD appear to represent distinct conditions with different molecular alterations and prognosis. They also pose different diagnostic challenges on biopsies and fine-needle aspiration (FNA) cytology. The aim of this study was to report on 3 cases of intra-abdominal RDD and perform an extensive review of the literature on FNA findings of RDD. MATERIALS AND METHODS: We reviewed FNA specimens from cases diagnosed histologically or cytologically as RDD during the past 10 years. We searched the PubMed and Google Scholar databases for cases of RDD sampled by FNA. RESULTS: We identified 3 cases of intra-abdominal RDD, involving the kidney, periportal lymph node, and pancreas. FNA of the latter was hypocellular with fibrosis and was nondiagnostic. FNA of the first 2 yielded hypercellular smears that were diagnosed as RDD due to the identification of emperipolesis occurring in large uni- or binucleated histiocytes with large nuclei, fine chromatin, and prominent nucleoli in smears and cell-block sections. Immunohistochemistry showed positive staining for S100 and CD68 and negative staining for CD1a. The large histiocytes with emperipolesis were more difficult to identify histologically and their demonstration required immunohistochemical stains. CONCLUSION: Our experience and an extensive review of the literature suggest that extranodal RDD can be diagnosed on FNA, and that the recognition of histiocytes with emperipolesis may be less challenging cytologically than histologically. The fibrosis frequently seen in extranodal RDD may lead to nondiagnostic aspirates, however.


Assuntos
Histiocitose Sinusal/diagnóstico , Rim/patologia , Linfonodos/patologia , Pâncreas/patologia , Doenças Raras/diagnóstico , Cavidade Abdominal , Adulto , Antígenos CD/imunologia , Antígenos CD1/imunologia , Antígenos de Diferenciação Mielomonocítica/imunologia , Biópsia por Agulha Fina , Emperipolese , Evolução Fatal , Feminino , Histiócitos/imunologia , Histiócitos/metabolismo , Histiocitose Sinusal/tratamento farmacológico , Humanos , Imuno-Histoquímica , Masculino , Pessoa de Meia-Idade , Doenças Raras/tratamento farmacológico , Proteínas S100/imunologia , Esteroides/uso terapêutico , Resultado do Tratamento , Adulto Jovem
20.
Elife ; 82019 05 01.
Artigo em Inglês | MEDLINE | ID: mdl-31042146

RESUMO

Bone marrow megakaryocytes engulf neutrophils in a phenomenon termed emperipolesis. We show here that emperipolesis is a dynamic process mediated actively by both lineages, in part through the ß2-integrin/ICAM-1/ezrin pathway. Tethered neutrophils enter in membrane-bound vesicles before penetrating into the megakaryocyte cytoplasm. Intracytoplasmic neutrophils develop membrane contiguity with the demarcation membrane system, thereby transferring membrane to the megakaryocyte and to daughter platelets. This phenomenon occurs in otherwise unmanipulated murine marrow in vivo, resulting in circulating platelets that bear membrane from non-megakaryocytic hematopoietic donors. Transit through megakaryocytes can be completed as rapidly as minutes, after which neutrophils egress intact. Emperipolesis is amplified in models of murine inflammation associated with platelet overproduction, contributing to platelet production in vitro and in vivo. These findings identify emperipolesis as a new cell-in-cell interaction that enables neutrophils and potentially other cells passing through the megakaryocyte cytoplasm to modulate the production and membrane content of platelets.


Assuntos
Plaquetas/metabolismo , Emperipolese/genética , Inflamação/genética , Megacariócitos/metabolismo , Animais , Células da Medula Óssea/metabolismo , Antígenos CD18/genética , Comunicação Celular , Citoplasma/genética , Citoplasma/metabolismo , Proteínas do Citoesqueleto/genética , Humanos , Inflamação/sangue , Inflamação/patologia , Molécula 1 de Adesão Intercelular/genética , Proteínas de Membrana Transportadoras/genética , Camundongos , Neutrófilos/metabolismo
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