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Journal articles on the topic 'Systemic EBV^+ T cell lymphoproliferative disease of childhood'

Author: Grafiati

Published: 4 June 2021

Last updated: 1 February 2022

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1

Quintanilla-Martinez, Leticia, Cecilia Ridaura, Florian Nagl, et al. "Hydroa vacciniforme-like lymphoma: a chronic EBV+ lymphoproliferative disorder with risk to develop a systemic lymphoma." Blood 122, no. 18 (2013): 3101–10. http://dx.doi.org/10.1182/blood-2013-05-502203.

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Key Points HVLL is a chronic EBV+ lymphoproliferative disorder of childhood with risk to develop systemic lymphoma. The disease shows favorable response to conservative therapy despite the presence of a T- or NK-cell monoclonal proliferation.

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2

Syrykh, Charlotte, Sarah Péricart, Claire Lamaison, Frédéric Escudié, Pierre Brousset, and Camille Laurent. "Epstein–Barr Virus-Associated T- and NK-Cell Lymphoproliferative Diseases: A Review of Clinical and Pathological Features." Cancers 13, no. 13 (2021): 3315. http://dx.doi.org/10.3390/cancers13133315.

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Epstein–Barr virus (EBV) is a ubiquitous virus detected in up to 95% of the general population. Most people are asymptomatic, while some may develop a wide range of EBV-associated lymphoproliferative disorders (LPD). Among them, EBV-positive T/NK LPD are uncommon diseases defined by the proliferation of T- or NK-cells infected by EBV. The 2017 World Health Organization (WHO) classification recognizes the following entities characterized by different outcomes: chronic active EBV infection of T- or NK-cell types (cutaneous and systemic forms), systemic EBV-positive T-cell lymphoma of childhood,

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3

Hyo, Rie, Yasunobu Abe, Kisato Nosaka, et al. "Proposal of Progressive Adult Onset EBV-Associated Lymphoproliferative Disorder (PAEBV) as a Rapid-Onset Neoplastic Disease." Blood 114, no. 22 (2009): 2920. http://dx.doi.org/10.1182/blood.v114.22.2920.2920.

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Abstract Abstract 2920 Poster Board II-896 [Background] Epstein-Barr virus (EBV) is currently recognized as a pathogen of several known human diseases ranging from transient infection, chronic lymphoproliferative disorder (LPD) to EBV-positive malignant lymphomas. Chronic active EBV infection (CAEBV) is typically an indolent but long-lasting EBV-LPD of childhood. Another well documented EBV-associated LPD is the post-transplant EBV-LPD, which also occurs in adults. Fulminant proliferation of EBV after primary infection or in the setting of CAEBV is an aggressive type of disease, which has rece

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4

Chen, Chien-Chin, Kung-Chao Chang, L. Jeffrey Medeiros, and Julia Yu-Yun Lee. "Hydroa Vacciniforme and Hydroa Vacciniforme-Like Lymphoproliferative Disorder: A Spectrum of Disease Phenotypes Associated with Ultraviolet Irradiation and Chronic Epstein–Barr Virus Infection." International Journal of Molecular Sciences 21, no. 23 (2020): 9314. http://dx.doi.org/10.3390/ijms21239314.

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Hydroa vacciniforme (HV) is a rare form of photosensitivity disorder in children and is frequently associated with Epstein–Barr virus (EBV) infection, whereas HV-like lymphoproliferative disorders (HVLPD) describe a spectrum of EBV-associated T-cell or natural killer (NK)-cell lymphoproliferations with HV-like cutaneous manifestations, including EBV-positive HV, atypical HV, and HV-like lymphoma. Classic HV occurs in childhood with papulovesicules on sun-exposed areas, which is usually induced by sunlight and ultraviolet irradiation, and mostly resolves by early adult life. Unlike classic HV,

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5

Chen, Zihang, Mi Wang, Pujun Guan, et al. "Comparison of Systemic EBV-positive T-Cell and NK-Cell Lymphoproliferative Diseases of Childhood Based on Classification Evolution." American Journal of Surgical Pathology 44, no. 8 (2020): 1061–72. http://dx.doi.org/10.1097/pas.0000000000001495.

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6

Hong, Mineui, Young Hyeh Ko, Keon Hee Yoo, et al. "EBV-Positive T/NK-Cell Lymphoproliferative Disease of Childhood." Korean Journal of Pathology 47, no. 2 (2013): 137. http://dx.doi.org/10.4132/koreanjpathol.2013.47.2.137.

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7

Yasa Benkli, C., A. Marcogliese, M. Elghetany, et al. "Diagnostic Challenges And Clinical Implications For Systemic EBV-Associated T-Cell Lymphoproliferative Disorders Of Childhood." American Journal of Clinical Pathology 154, Supplement_1 (2020): S158. http://dx.doi.org/10.1093/ajcp/aqaa161.345.

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Abstract Introduction/Objective Systemic EBV-associated T-cell lymphoproliferative disorders of childhood (S-EBV-T-LPD) comprise three major forms: EBV-positive hemophagocytic lymphohistiocytosis (EBV-HLH), systemic EBV-positive T- cell lymphoma (S-EBV-TCL), and systemic T-cell chronic active EBV infection (S-T-CAEBV). These disorders are rare in children and young adults in Western countries and are associated with poor outcomes. Frequently patients were treated initially for EBV-HLH and subsequently found to have relapsed/refractory EBV-HLH vs S-EBV-TCL or overt EBV+ TCL, the latter of which

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8

Dutz, Jan P., Loralyn Benoit, Xiaoxia Wang, et al. "Lymphocytic vasculitis in X-linked lymphoproliferative disease." Blood 97, no. 1 (2001): 95–100. http://dx.doi.org/10.1182/blood.v97.1.95.

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Abstract Systemic vasculitis is an uncommon manifestation of X-linked lymphoproliferative disease (XLP), a disorder in which there is a selective immune deficiency to Epstein-Barr virus (EBV). The molecular basis for XLP has recently been ascribed to mutations within SLAM-associated protein (SAP), an SH2 domain–containing protein expressed primarily in T cells. The authors describe a patient who died as a result of chronic systemic vasculitis and fulfilled clinical criteria for the diagnosis of XLP. Sequencing of this patient'sSAP gene uncovered a novel point mutation affecting the SH2 domain.

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9

Zhou, Jiehao, Dehua Wang, and Mehdi Nassiri. "Clonal CD8+ T Lymphocytic Proliferation and Karyotypical Abnormalities in an EBV Associated Hemophagocytic Lymphohistiocytosis." Case Reports in Pathology 2015 (2015): 1–6. http://dx.doi.org/10.1155/2015/513968.

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EBV associated hemophagocytic lymphohistiocytosis and EBV-positive T cell lymphoproliferative disease of childhood share many histologic and clinical features, which sometimes makes it very difficult to render a definitive diagnosis. In this report, we present a 16-year-old male who developed symptoms clinically consistent with EBV associated hematophagocytic lymphohistiocytosis including fulfilling most of HLH diagnostic criteria and responding promptly to HLH targeted therapy. However, histologic and cytogenetics features of this case are very concerning for EBV-positive T cell lymphoprolife

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10

Kimura, Hiroshi, Yoshinori Ito, Shinji Kawabe, et al. "EBV-associated T/NK–cell lymphoproliferative diseases in nonimmunocompromised hosts: prospective analysis of 108 cases." Blood 119, no. 3 (2012): 673–86. http://dx.doi.org/10.1182/blood-2011-10-381921.

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AbstractEBV-associated T/NK–cell lymphoproliferative disease (T/NK-LPD) is defined as a systemic illness characterized by clonal proliferation of EBV-infected T or NK cells. We prospectively enrolled 108 nonimmunocompromised patients with this disease (50 men and 58 women; median onset age, 8 years; age range, 1-50 years) evidenced by expansion of EBV+ T/NK cells in the peripheral blood; these were of the T-cell type in 64 cases and of the NK-cell type in 44, and were clinically categorized into 4 groups: 80 cases of chronic active EBV disease, 15 of EBV-associated hemophagocytic lymphohistioc

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11

Leeborg, Nicky, Thomas Russell, and Guang Fan. "Systemic Epstein-Barr Virus–Positive T-Cell Lymphoproliferative Disease of Childhood." Pathology Case Reviews 17, no. 3 (2012): 120–24. http://dx.doi.org/10.1097/pcr.0b013e318258eeba.

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12

Mun, J., K. Hur, and B. Han. "507 Hydroa vacciniforme-like lymphoproliferative disorder and systemic EBV+ T cell lymphoma of childhood and adulthood." Journal of Investigative Dermatology 139, no. 9 (2019): S301. http://dx.doi.org/10.1016/j.jid.2019.07.557.

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13

Qian, Meihua, Wensong Wang, Manling Wang, et al. "The Treatment of ECVP Plus G-Csf on Epstein–Barr Virus-Associated T Lymphoproliferative Disease (EBV+ T-LPD) in Adult." Blood 118, no. 21 (2011): 4384. http://dx.doi.org/10.1182/blood.v118.21.4384.4384.

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Abstract Abstract 4384 Epstein–Barr virus-associated T lymphoproliferative disease (EBV+ T-LPD, non-immunocompromised hosts) is rare on adults. Normally it is only found on children. This disease, according to recent pathological categorization, can be categorized into following subtypes: (i) category A1, polymorphic LPD without clonal proliferation of EBV-infected cells; (ii) category A2, polymorphic LPD with clonality; (iii) category A3, monomorphic LPD (T-cell or NK cell lymphoma/leukemia) with clonality; and (iv) category B, monomorphic LPD (T-cell lymphoma) with clonality and fulminant cl

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14

Harker-Murray, P. D., V. Dayton, J. Neglia, and J. Tolar. "Treatment of EBV-associated T cell post-transplant lymphoproliferative disorder with CNS involvement in a pediatric solid-organ transplant patient." Journal of Clinical Oncology 24, no. 18_suppl (2006): 9040. http://dx.doi.org/10.1200/jco.2006.24.18_suppl.9040.

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9040 Background: Post-transplant lymphoproliferative disorder (PTLD) is a known complication of immunosuppression following solid organ and stem cell transplantation. It ranges from benign lymphoid hyperplasia to fulminant systemic disease with high mortality. Most cases are B lineage and associated with Epstein-Barr virus (EBV+). T cell PTLD is rare and usually EBV negative. To date only 5 cases of EBV+ T cell PTLD have been reported in pediatric patients and none have had documented involvement of the central nervous system (CNS). Methods: We provide clinical, histologic, immunophenotypic an

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15

Cohen, Jeffrey I., Irini Manoli, Kennichi Dowdell, et al. "Hydroa vacciniforme–like lymphoproliferative disorder: an EBV disease with a low risk of systemic illness in whites." Blood 133, no. 26 (2019): 2753–64. http://dx.doi.org/10.1182/blood.2018893750.

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AbstractPatients with classic hydroa vacciniforme–like lymphoproliferative disorder (HVLPD) typically have high levels of Epstein-Barr virus (EBV) DNA in T cells and/or natural killer (NK) cells in blood and skin lesions induced by sun exposure that are infiltrated with EBV-infected lymphocytes. HVLPD is very rare in the United States and Europe but more common in Asia and South America. The disease can progress to a systemic form that may result in fatal lymphoma. We report our 11-year experience with 16 HVLPD patients from the United States and England and found that whites were less likely

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16

Prockop, Susan, Stephanie Suser, Ekaterina Doubrovina, et al. "Adoptive Therapy with EBV-Specific T Cells for Treatment of CNS EBV Post-Transplant Lymphoproliferative Disease Arising after Hematopoietic Stem Cell Transplant or Solid Organ Transplant." Blood 132, Supplement 1 (2018): 4590. http://dx.doi.org/10.1182/blood-2018-99-120133.

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Abstract Adoptive immunotherapy with EBV-specific T-cells (EBV-CTLs) derived from primary hematopoietic transplant donors is effective in the treatment of EBV disease complicating allogeneic hematopoietic stem cell transplant (HCT). In addition, third party donor-derived EBV-CTLs (tabelecleucel) have demonstrated efficacy in the treatment of EBV post-transplant lymphoproliferative in the setting of both HCT and solid organ transplant (SOT). While the introduction of first line therapy with rituximab has reduced the mortality associated with EBV-PTLD, EBV PTLD involving the central nervous syst

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17

Crawford, Dorothy H. "Biology and disease associations of Epstein–Barr virus." Philosophical Transactions of the Royal Society of London. Series B: Biological Sciences 356, no. 1408 (2001): 461–73. http://dx.doi.org/10.1098/rstb.2000.0783.

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Epstein–Barr virus (EBV) is a human herpesvirus which infects almost all of the world's population subclinically during childhood and thereafter remains in the body for life. The virus colonizes antibody–producing (B) cells, which, as relatively long–lived resting cells, are an ideal site for long–term residence. Here EBV evades recognition and destruction by cytotoxic T cells. EBV is passed to naive hosts in saliva, but how the virus gains access to this route of transmission is not entirely clear. EBV carries a set of latent genes that, when expressed in resting B cells, induce cell prolifer

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18

Silins, Sharon L., Martina A. Sherritt, Jodie M. Silleri, et al. "Asymptomatic primary Epstein-Barr virus infection occurs in the absence of blood T-cell repertoire perturbations despite high levels of systemic viral load." Blood 98, no. 13 (2001): 3739–44. http://dx.doi.org/10.1182/blood.v98.13.3739.

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Abstract Primary infection with the human herpesvirus, Epstein-Barr virus (EBV), may result in subclinical seroconversion or may appear as infectious mononucleosis (IM), a lymphoproliferative disease of variable severity. Why primary infection manifests differently between patients is unknown, and, given the difficulties in identifying donors undergoing silent seroconversion, little information has been reported. However, a longstanding assumption has been held that IM represents an exaggerated form of the virologic and immunologic events of asymptomatic infection. T-cell receptor (TCR) repert

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19

Kim, Do-Hoon, Myungshin Kim, Yonggoo Kim, et al. "Systemic Epstein-Barr Virus-positive T-Cell Lymphoproliferative Disease of Childhood With Good Response to Steroid Therapy." Journal of Pediatric Hematology/Oncology 39, no. 8 (2017): e497-e500. http://dx.doi.org/10.1097/mph.0000000000000909.

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20

Paisiou, A., C. M. Vadikolia, K. Stefanaki, et al. "EBV Related Lymphoproliferative Disorders Post Allogeneic Bone Marrow Transplantation for Haematological Malignancies." Blood 126, no. 23 (2015): 5480. http://dx.doi.org/10.1182/blood.v126.23.5480.5480.

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Abstract Background: Post transplant lymphoproliferative disorder (PTLD) is a heterogeneous complication of HSCT. It comprises a spectrum of pathogenetic mechanisms and clinical manifestations. It is mostly associated with EBV infection, either as a consequence of reactivation in the post transplant period or less frequently from primary infection. WHO classification defines four major histopathologic subtypes: 1. early plasmacytic hyperplasia and infectious mononucleosis (IM)-like 2. polymorphic lesions which may be polyclonal or focally monoclonal (P-PTLD) 3. monomorphic lesions that fulfill

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21

Yang, Naery, Wha Soon Chung, Yeung Chul Mun, and Jungwon Huh. "Systemic Epstein-Barr Virus-Positive T-cell Lymphoproliferative Disease of Childhood Presenting as Hemophagocytic Lymphohistiocytosis with Chromosomal Abnormalities." Journal of Laboratory Medicine and Quality Assurance 36, no. 4 (2014): 210–15. http://dx.doi.org/10.15263/jlmqa.2014.36.4.210.

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22

Savasan, Sureyya, Batool Al-Qanber, Steven Buck, Erin Wakeling, and Manisha Gadgeel. "Clonal T-Large Granular Lymphocyte Proliferations in Childhood: Friend or Foe?" Blood 132, Supplement 1 (2018): 3719. http://dx.doi.org/10.1182/blood-2018-99-119124.

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Abstract Background: The pathophysiology of clonal T-large granular lymphocyte (T-LGL) proliferations is not well understood; the distinction between reactive and malignant entities is not very clear given the fact that acquired STAT3 mutations can be seen with clonal T-LGL proliferations in non-malignant Felty syndrome and with frequent use of immunosuppression for the treatment of T-LGL leukemia. Historically, determination of clonality has been often seen as an indicator of T-LGL leukemia. We reviewed our experience on clonal T-LGL proliferations in children. Material and Methods: T-LGLs we

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23

Kulm, Elaine, Sharon Webster, Kate Howe, Amy Rump, Gulbu Uzel, and V. Koneti Rao. "Autoimmune Cytopenias in Nonmalignant Lymphoproliferative Disorders: Thinking Beyond ALPS(Autoimmune Lymphoproliferative Syndrome)." Blood 136, Supplement 1 (2020): 48. http://dx.doi.org/10.1182/blood-2020-137640.

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The ALPS Clinic at NIH has studied autoimmune lymphoproliferative disorders for over 30 years elucidating the genetic underpinnings and natural history of Autoimmune Lymphoproliferative Syndrome (ALPS) due to FAS gene defects (Blood 2014). Over the years, we continue to receive and work-up numerous referrals for diseases that masquerade like ALPS-FAS, presenting with multi-lineage cytopenias due to autoimmune peripheral destruction and splenic sequestration in the setting of non-malignant lymphoproliferation. In a review of 259 cases evaluated by our team, there were 150 ALPS-FAS, 54 Activated

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24

YOSHII, MIYUKI, MITSUAKI ISHIDA, KEIKO HODOHARA, et al. "Systemic Epstein-Barr virus-positive T-cell lymphoproliferative disease of childhood: Report of a case with review of the literature." Oncology Letters 4, no. 3 (2012): 381–84. http://dx.doi.org/10.3892/ol.2012.754.

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25

Crane, Genevieve Marie, Helen Powell, Rumen Kostadinov, et al. "Lymphoproliferative Disease Risk in Patients with Autoimmune Disease: Clustering of Primary CNS Lymphoma with Drug Regimen and Disease Process." Blood 126, no. 23 (2015): 1490. http://dx.doi.org/10.1182/blood.v126.23.1490.1490.

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Abstract Introduction: While immunosuppression for solid organ transplant is associated with an increased risk of lymphoproliferative disease (LPD), this has been more difficult to establish in autoimmune disorders, even though patients are often treated with similar agents. One reason is that autoimmune disease may elevate baseline LPD risk. However, associations have been shown with certain rare types of LPD; most strikingly hepatosplenic lymphoma, now known to occur as a consequence of anti-TNF-alpha therapy in young men with inflammatory bowel disease (IBD) (J Ped Gast Nutr. 2007; 44:265-7

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26

Sekiguchi, Nodoka, Kazuyuki Matsuda, Kayoko Momose, Hideki Makishima, Toshiro Ito, and Fumihiro Ishida. "STAT3 Gene Mutations and the Association with Pure Red Cell Aplasia in Large Granular Lymphocyte Leukemia." Blood 120, no. 21 (2012): 2668. http://dx.doi.org/10.1182/blood.v120.21.2668.2668.

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Abstract Abstract 2668 Large granular lymphocyte leukemia (LGL-L) is a proliferative disorder of cytotoxic T cells or NK cells frequently complicated with cytopenia and autoimmune phenomena. In the current WHO classification, T-cell large granular lymphocyte leukemia (T LGL-L), and chronic lymphoproliferative disorders of NK cells (CLPD-NK) are included in this category. Aggressive NK cell leukemia (ANKL) and Epstein-Barr virus (EBV)-positive T-cell lymphoproliferative disease of childhood (EBV-T LPD) are also categorized as entities in the classification based on the clinical characteristics

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27

Yonese, Ichiro, Chizuko Sakashita, Ken-Ichi Imadome, et al. "Nationwide survey of systemic chronic active EBV infection in Japan in accordance with the new WHO classification." Blood Advances 4, no. 13 (2020): 2918–26. http://dx.doi.org/10.1182/bloodadvances.2020001451.

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Abstract Systemic chronic active Epstein-Barr virus infection (sCAEBV) was defined as a T- or NK-cell neoplasm in the 2017 World Health Organization (WHO) classification. To clarify the clinical features of sCAEBV under this classification and review the effects of chemotherapy, we performed a nationwide survey in Japan from 2016 through 2018 of patients with sCAEBV newly diagnosed from January 2003 through March 2016. One hundred cases were evaluated. The patients were aged 1 to 78 years (median, 21) and included 53 males and 47 females. Spontaneous regression was not observed in patients wit

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28

Jost, Philipp J., Monica Yabal, Heiko Adler, et al. "A Mouse Model for XLP-2 Disease Uncovers a Critical Function for IL-1beta and TNF in Driving Hyper-Inflammation." Blood 124, no. 21 (2014): 1403. http://dx.doi.org/10.1182/blood.v124.21.1403.1403.

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Abstract The hyper-inflammatory syndrome X-linked lymphoproliferative syndrome type 2 (XLP-2) is defined by mutations in BIRC4 (XIAP). XLP-2 is often diagnosed in paediatric patients and is characterized by hyper-inflammation triggered by common viral infections. Symptoms include splenomegaly, HLH, fevers, and chronic haemorrhagic colitis among others. Recent work has also shown that mutations in BIRC4 predispose to the development of early-onset IBD, which is not necessarily associated with symptoms of systemic hyper-inflammation. Symptoms of XLP-2 are mostly attributed to the aberrant activa

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29

Fekrazad, M. Houman, Lizabeth Rosenbaum, Mohammad A. Vasef, and Ian Rabinowitz. "EBV-Associated Follicular Dendritic Cell Tumor of the Spleen." Blood 110, no. 11 (2007): 4509. http://dx.doi.org/10.1182/blood.v110.11.4509.4509.

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Abstract Epstein-Barr virus (EBV) expressing follicular dendritic cell proliferations in the spleen is an extremely rare but distinct variant of follicular dendritic cell tumor (FDCT). This neoplasm typically presents as an intra-abdominal mass with a predilection to involve spleen or liver with histologic features reminiscent of inflammatory pseudotumor (IP). However, in contrast to IP, this distinct FDCT consistently is associated with EBV. A subset of splenic tumors previously reported as examples of IP or inflammatory myofibroblastic tumor represent EBV-associated FDCT retrospectively. Des

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30

Tacyildiz, Nurdan, Gulsah Tanyildiz, Gulsan Yavuz, et al. "Lymphoma Patients Secondary to Congenital and Acquired Immunodeficiency Syndroms in a Turkish Pediatric Oncology Center." Blood 124, no. 21 (2014): 5445. http://dx.doi.org/10.1182/blood.v124.21.5445.5445.

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Abstract PURPOSE An increased incidence of lymphoma is seen in various types of immune deficiency syndromes,including congenital immune deficiency diseases, organ transplantation with iatrogenic immunosuppression and autoimmune disorders. Prognosis of the lymphomas secondary to immunodeficiencies is stil poor. We aimed to analyse clinical features and treatment results of our patients that diagnosed as lymphoma and have immundeficiency syndrom. PATIENTS Between 2002-2014, we have seen 12 (7male, 5 female) childhood lymphoma that related immunodeficiencies. Ages of patients were between 4-15 ye

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31

Brunet, Vanessa, Michel Pavic, Sofia Marouan, Isabelle Fleury, and Jean-Francois Castilloux. "Literature Review of All Cases of Aggressive T-Cell Large Granular Lymphocytic Leukemia Cases and Report of an Additional Case." Blood 132, Supplement 1 (2018): 5421. http://dx.doi.org/10.1182/blood-2018-99-111726.

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Abstract Large granular lymphocyte (LGL) leukemia is a spectrum of rare lymphoproliferative disorders, classified into T-cell LGL leukemia, chronic lymphoproliferative disorder of NK-cells and aggressive NK-cell leukemia; chronic NK-cell leukemia is a provisional diagnosis. However, we identified fourteen cases of aggressive T-LGL leukemia retrieved in the literature. Considering this unusual and rare clinical presentation, we are reporting a literature review and presenting an additional case. Leukemic cells of T-LGL leukemia have a characteristic phenotype (CD3+CD8+CD16+CD57+) and show clona

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32

Kaji, Daisuke, Manabu Kusakabe, Mamiko Sakata-Yanagimoto, et al. "Clinicopathological Analysis of "Other Iatrogenic Immunodeficiency-Associated Lymphoproliferative Disorders" Reveals a Favorable Outcome Independent of the Effectiveness of Methotrexate Discontinuation in Autoimmune Disease Patients." Blood 134, Supplement_1 (2019): 4128. http://dx.doi.org/10.1182/blood-2019-127521.

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[Background] Patients treated with immunosuppressive drugs for autoimmune diseases may suffer "other iatrogenic immunodeficiency-associated lymphoproliferative disorders" (OIIA-LPD). Some OIIA-LPDs regress after drug withdrawal but others need cytotoxic chemotherapy. However, the factors associated with a response to drug cessation remain unknown. [Methods] We collected clinical data on OIIA-LPD diagnosed between 2009 and 2018 at the University of Tsukuba Hospital and Toranomon Hospital in Japan. Clinicopathological features were retrospectively analyzed. The probability of overall survival (O

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33

Buckley, Sarah A., Daniel Egan, and Roland B. Walter. "Hemophagocytic Lymphohistiocytosis in Adults: A Case Series of 30 Patients At a U.S. Tertiary Care Center." Blood 120, no. 21 (2012): 4681. http://dx.doi.org/10.1182/blood.v120.21.4681.4681.

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Abstract Abstract 4681 Background: Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening disorder characterized by overwhelming immune activation that results in excessive inflammation, phagocytosis of blood cells by macrophages, and end-organ damage. Both familial forms (“primary HLH”), often presenting in childhood, and sporadic forms (“secondary HLH”), often presenting in adults, have been recognized. While pediatric HLH is well characterized, only limited data is available on the diagnosis and treatment of HLH in adults. This study, the largest single center case series of adult H

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34

Constantine, Gregory, Helen Su, Les Folio, Joshua Milner, and V. Koneti Rao. "Clinical Spectrum of Patients with Pathogenic Variant of STAT3 conferring Gain-of-Function: A Mimic of Autoimmune Lymphoproliferative Syndrome." Blood 132, Supplement 1 (2018): 3723. http://dx.doi.org/10.1182/blood-2018-99-111950.

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Abstract Autoimmune lymphoproliferative syndrome (ALPS) is a rare genetic disorder caused by defective fas-mediated apoptosis. Patients often present in childhood with lymphoproliferation, splenomegaly and multilineage cytopenias (Price et al. Natural history of autoimmune lymphoproliferative syndrome associated with FAS gene mutations. Blood 2014). Though mutations in the FAS gene account for the majority of cases, an estimated 20% of patients who have no defined genetic cause are classified as ALPS-U (Shah et al. Autoimmune lymphoproliferative syndrome: an update and review of the literature

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35

Grimes, Amanda Bell, Taylor Kim, Susan E. Kirk, Michele P. Lambert, Rachael F. Grace, and Jenny M. Despotovic. "Targeting Immune Dysregulation in Childhood Evans Syndrome." Blood 132, Supplement 1 (2018): 3564. http://dx.doi.org/10.1182/blood-2018-99-110734.

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Abstract Background: Evans Syndrome (ES) describes the simultaneous or sequential occurrence of 2 or more autoimmune cytopenias - most often autoimmune hemolytic anemia (AIHA) and immune thrombocytopenia (ITP), but also autoimmune neutropenia (AIN) - which are often refractory to therapy and chronic in nature. Historically, ES has been attributed to idiopathic autoantibody production, but recent advances in our understanding of this disease have revealed associations with more well-described underlying disorders of immune regulation when properly investigated. These include autoimmune lymphopr

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36

Arai, Ayako, Ichiro Yonese, Chizuko Sakashita, et al. "A Nationwide Survey on Chronic Active Epstein-Barr Virus Infection in Japan Based on New Diagnostic Criteria: Clinical Features and Treatment Strategies." Blood 132, Supplement 1 (2018): 1631. http://dx.doi.org/10.1182/blood-2018-99-117854.

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Abstract Background and aims Chronic active Epstein-Barr virus infection (CAEBV) is classified into T- or NK-cell neoplasms in the new WHO classification revised in 2017. Allogeneic stem cell transplantation (allo-HSCT) has recently been reported to be an effective treatment for this disorder. Conversely, effects of chemotherapies on CAEBV have not yet been examined in a large number of patients. To clarify clinical features and the current state of chemotherapies for CAEBV under the new definition of the disease, we performed a nationwide survey in Japan. Methods Questionnaires were sent to a

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Kazi, Sajida, Abhinav Mathur, Gwen M. Wilkie, et al. "Outcomes after Third Party, Viral-Specific, Cytotoxic Lymphocytes for Immunosuppression- and Epstein-Barr Virus-Associated Lymphoproliferative Disease." Blood 132, Supplement 1 (2018): 3329. http://dx.doi.org/10.1182/blood-2018-99-114477.

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Abstract Epstein-Barr virus associated lymphoproliferative disease is a serious complication of immunosuppression, particularly after transplantation. Initial treatments usually comprise reduction of immunosuppression, rituximab and/or chemotherapy. However, some patients fail to respond or are unsuitable for chemotherapy and so are candidates for adoptive cellular therapy. Here we report outcomes from the use of a bank of 25 third party derived Epstein-Barr virus specific lymphocyte cell lines cryopreserved for immediate use, issued on a best-HLA match basis. Cells have been issued to 70 pati

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M, Vinodhini, Anant Gokarn, Sachin Punatar, et al. "Incidence, Risk Factors, and Outcomes of Viral Reactivations in Alternative Donor Haematopoietic Stem Cell Transplant." Blood 136, Supplement 1 (2020): 24–25. http://dx.doi.org/10.1182/blood-2020-141751.

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Introduction : There has been an increase in number of alternative donor [Unrelated donor (UD) or Haploidentical (HIT)] transplants in recent years due to better graft vs. host disease (GVHD) prophylaxis and comparable outcomes with matched sibling transplants. However, viral reactivations frequently complicate these transplants and can lead to organ dysfunction, poor graft function and death. We aimed to analyze incidence, risk factors, and outcomes of viral reactivations in alternative donor transplants at our centre. Methods: All UD or HIT transplants performed between January 1st 2009 and

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Kakinoki, Yasutaka, Satomi Matsuoka, Junichi Hashiguchi, Koji Chiba, and Takayoshi Miyake. "Successful treatment of immediate allogeneic myeloablative hematopoietic stem cell transplantation from a HLA ‐mismatched sibling donor for active systemic epstein–barr virus‐positive T‐cell lymphoproliferative disease of childhood following primary acute epstein–barr virus infection." Clinical Case Reports 3, no. 4 (2015): 231–36. http://dx.doi.org/10.1002/ccr3.204.

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Moser, Olga, Arnaud Dalissier, Eric Beohou, et al. "Stem Cell Transplantation for Pediatric Patients with Non-Anaplastic Peripheral T-Cell Lymphoma on Behalf of the EBMT-Pediatric Diseases Working Party." Blood 132, Supplement 1 (2018): 5787. http://dx.doi.org/10.1182/blood-2018-99-116809.

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Abstract Peripheral T-cell lymphomas (PTCL) other than anaplastic large cell lymphoma are rare in children, and data about outcome and treatment results especially regarding the role of stem cell transplantation (SCT) are scarce. Here we present the results of a retrospective study of SCT for pediatric patients with PTCL within the European Group of Bone marrow transplantation (EBMT). Out of 125 patients aged <18 years with PTCL diagnosed between 1995 and 2015 who were registered to the EBMT database, reports about the primary therapy and data about the course of SCT were sufficient for ana

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Solomon, Scott R., Xu Zhang, Melhem Solh, Lawrence E. Morris, H. Kent Holland, and Asad Bashey. "Long-Term Follow-up of Myeloablative Haploidentical (HID) Peripheral Blood Stem Cell Transplantation and Post-Transplant Cyclophosphamide (PTCy) Following Fludarabine and Total Body Irradiation (TBI)." Blood 132, Supplement 1 (2018): 3368. http://dx.doi.org/10.1182/blood-2018-99-111029.

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Abstract Haploidentical donor (HID) transplant with post-transplant cyclophosphamide (PTCy) is being increasingly utilized worldwide. Although the original "Baltimore" protocol employed nonmyeloablative conditioning, there has been increasing interest in the use of myeloablative conditioning (MAC) in an attempt to decrease the risk of disease recurrence and graft failure. We present the long-term follow-up results of 82 consecutive patients receiving a MAC HID transplant at a single institution between 2011-2017, following a uniform protocol of Fludarabine/total body irradiation (TBI) 1200cGy

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Deaver, Darcie, Mojdeh Naghashpour, and Lubomir Sokol. "Kikuchi-Fujimoto Disease In the United States: Clinical and Laboratory Characteristics and Outcomes." Blood 116, no. 21 (2010): 5100. http://dx.doi.org/10.1182/blood.v116.21.5100.5100.

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Abstract Abstract 5100 Introduction: Kikuchi-Fujimoto Disease (KFD), also known as histiocytic necrotizing lymphadenitis, is a benign, idiopathic, self-limiting disease manifesting primarily as unilateral cervical lymphadenopathy in younger Asian women. In western countries this condition is very rare. Objectives: To compare clinical laboratory characteristics and delineate major differences between KFD and malignant lymphoproliferative disorders. Methods: This is a retrospective review of two patients with KFD that were evaluated in our institution between 2008 and 2010. Results: Two previous

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Asano, Naoko, Jun-Ichi Tamaru, Fumihiro Ishida, et al. "Cytotoxic Molecule (CM)-Positive Classical Hodgkin Lymphoma: a Clinicopathologic Study in Comparison with Nodal Peripheral T-Cell Lymphoma of Not Otherwise Specified Type Possessing CM Expression." Blood 114, no. 22 (2009): 1549. http://dx.doi.org/10.1182/blood.v114.22.1549.1549.

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Abstract Abstract 1549 Poster Board I-572 Classical Hodgkin lymphoma (CHL) is characterized by Hodgkin and Reed Sternberg (H-RS) cells, which are B-cell origin in many cases. Recently, we highlighted an adverse prognostic significance of cytotoxic molecule (CM) expression among CHL patients (Asano N, et al. J Clin Oncol. 2006). However, the clinical characteristics of CM-positive CHL still remain controversial. We here document the clinicopathologic profiles of 35 patients with CM-positive CHL, consisting of 23 men and 12 women with a median age of 50 years (range, 16 - 84 years). All patients

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Hamdi, Leila, Rita Creidy, Patricia Rince, et al. "A Study of Immune Deficiency as Risk Factor of Hodgkin's Lymphoma in Children." Blood 114, no. 22 (2009): 1559. http://dx.doi.org/10.1182/blood.v114.22.1559.1559.

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Abstract Abstract 1559 Poster Board I-582 Introduction Median age of Hodgkin's Lymphoma (HL) cases in children is 12-14 years. Epidemiological and histological features are similar to HL occurring in young adults. In contrast, HL under the age of 10 has specific features; it predominates strongly in boys and it is more frequently associated with Epstein-Barr virus (EBV). There is accumulating evidence for an inherited susceptibility to HL based on many reports of familial aggregation of the disease in adults and in childhood. Cohort studies have shown that patients affected by several immune d

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Schram, Alison M., Ann Mullally, Annemarie E. Fogerty, Elena Massarotti, and Nancy Berliner. "Hemophagocytic Lymphohistiocytosis: The Partners Healthcare Experience over the Past 8 Years." Blood 124, no. 21 (2014): 4104. http://dx.doi.org/10.1182/blood.v124.21.4104.4104.

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Abstract Introduction Hemophagocytic Lymphohistiocytosis (HLH) is a rare disorder of uncontrolled immune activation characterized by clinical and laboratory evidence of severe inflammation. This syndrome can be caused by genetic mutations affecting cytotoxic function (familial HLH) or secondary to infectious, rheumatologic, malignant, or metabolic conditions (acquired HLH). Familial HLH typically presents in childhood and is well described. Less is known about the clinical characteristics, appropriate diagnostic criteria, and optimal treatment in the adult population. The purpose of this study

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Otrock, Zaher K., and Charles S. Eby. "Spectrum Of Clinical and Laboratory Findings In Adult Patients With Hemophagocytic Lymphohistiocytosis: Significance Of Ferritin Levels." Blood 122, no. 21 (2013): 1037. http://dx.doi.org/10.1182/blood.v122.21.1037.1037.

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Abstract Background Hemophagocytic lymphohistiocytosis(HLH) is a rare frequently fatal group of immunodeficiencies that mostly present with cytopenias and infections. It is characterized by an immune dysregulation that results in excessive cytokine expression, phagocytosis of blood cells by activated macrophages, and end-organ damage. There are familial forms (“primary HLH”), typically presenting in childhood, and sporadic forms (“secondary HLH”), often presenting in adults. While primary HLH is often limited to mutations affecting perforin dependent cytotoxicity, limited data are available on

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Han, Byeol, Keunyoung Hur, Jungyoon Ohn, et al. "Hydroa vacciniforme-like lymphoproliferative disorder in Korea." Scientific Reports 10, no. 1 (2020). http://dx.doi.org/10.1038/s41598-020-76345-2.

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Abstract Hydroa vacciniforme-like lymphoproliferative disorder (HVLPD) is a rare Epstein–Barr virus (EBV)-associated lymphoproliferative disease. The disease course of HVLPD varies from an indolent course to progression to aggressive lymphoma. We investigated the characteristics of HVLPD in Korean patients. HVLPD patients at Seoul National University Hospital between 1988 and 2019 were retrospectively analyzed. This study included 26 HVLPD patients who all presented with recurrent papulovesicular and necrotic eruption on the face, neck, and extremities. EBV was detected from the skin tissues o

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Wang, Ziyao, Shoichi Kimura, Hiromi Iwasaki, et al. "Clinicopathological findings of systemic Epstein-Barr virus-positive T-lymphoproliferative diseases in younger and older adults." Diagnostic Pathology 16, no. 1 (2021). http://dx.doi.org/10.1186/s13000-021-01107-1.

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Abstract Background Systemic Epstein-Barr virus+ T-cell lymphoma (sEBV+ TCL) occurs in childhood and young adults, and is exceptionally rare in older adults. Methods We investigated clinicopathological features in 16 patients of various ages with systemic EBV+ CD8+ T-lymphoproliferative diseases. Results Eight younger patients and four of eight older adults had sEBV+ CD8+ TCL, with invasion by medium-sized to/or large atypical lymphocytes primarily in bone marrow and lymph nodes, hemophagocytic lymphohistiocytosis (HLH), and progressive clinicopathological course. A further two patients demons

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Xiao, Shu-Yuan. "Comparison of Systemic EBV-positive T-Cell and NK-Cell Lymphoproliferative Diseases (LPD) of Childhood Based on Classification Evolution." American Journal of Surgical Pathology Publish Ahead of Print (December 22, 2020). http://dx.doi.org/10.1097/pas.0000000000001652.

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Volk, Valery, Sebastian J. Theobald, Simon Danisch, et al. "PD-1 Blockade Aggravates Epstein–Barr Virus+ Post-Transplant Lymphoproliferative Disorder in Humanized Mice Resulting in Central Nervous System Involvement and CD4+ T Cell Dysregulations." Frontiers in Oncology 10 (January 12, 2021). http://dx.doi.org/10.3389/fonc.2020.614876.

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Post-transplant lymphoproliferative disorder (PTLD) is one of the most common malignancies after solid organ or allogeneic stem cell transplantation. Most PTLD cases are B cell neoplasias carrying Epstein-Barr virus (EBV). A therapeutic approach is reduction of immunosuppression to allow T cells to develop and combat EBV. If this is not effective, approaches include immunotherapies such as monoclonal antibodies targeting CD20 and adoptive T cells. Immune checkpoint inhibition (ICI) to treat EBV+ PTLD was not established clinically due to the risks of organ rejection and graft-versus-host disea

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