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Seung Hwan Moon, Ah Young Lee, Won-Seog Kim, Seok Jin Kim, Young Seok Cho, Yearn Seong Choe, Byung-Tae Kim, Kyung-Han Lee
Subjects were 45 patients with angioimmunoblastic T-cell lymphoma (AITL) who underwent 2-[(18)F]-fluoro-2-deoxy-d-glucose (FDG) positron-emission tomography/computed tomography (PET/CT) at baseline and interim after 2-4 cycles. Predictors of progression-free survival (PFS) and overall survival (OS) were assessed. Positive interim PET/CT (Deauville score ≥3) was a significant independent predictor of poor PFS (Hazard ratio, 4.42; p=.028), and showed marginal significance to predict OS (p=.065). Less than 60% decrease in the average change of maximum standardized uptake value normalized by lean body mass (SULmax) also was a significant independent predictor of poor PFS (Hazard ratio, 12...
October 9, 2016: Leukemia & Lymphoma
Ya-Ting Yang, Cheng-Jeng Tai, Chiehfeng Chen, Hong-Cheng Wu, Natalia Mikhaylichenko, Hsien-Tsai Chiu, Yun-Yi Chen, Yi-Hsin Elsa Hsu
BACKGROUND: The goal of this study was to perform a systematic review to examine the efficacy and safety of various salvage therapy regimens on patients with relapsed/refractory PTCL. METHOD: The electronic searches were performed using PubMed, Cochrane Library, EMBASE, and Web of Science from inception through June 2015, with search terms related to relapsed/refractory PTCL, salvage chemotherapy regimens, and clinical trials. An eligible study met the following inclusion criteria: (1) Patients had refractory or relapsed PTCL; (2) drug regimens were used for salvage therapy; (3) the study was a clinical trial; (4) the study reported on a series of at least 10 patients of PTCL...
2016: PloS One
Claudius Irlé, Jonathan Weintraub
Peripheral T-cell lymphomas (PTCLs) are a heterogeneous group of aggressive non-Hodgkin lymphomas. Angioimmunoblastic T-cell lymphoma (AITL) is a common subtype of PTCL, and patients with AITL typically have a poor prognosis with limited treatment options. Clinical studies have demonstrated the activity of romidepsin, a structurally unique, potent, bicyclic class 1 selective histone deacetylase inhibitor, in patients with relapsed or refractory AITL. In the case presented herein, we describe a patient treated with single-agent romidepsin at first diagnosis of AITL, resulting in complete remission for over 2 years and leading to the use of maintenance dosing...
2016: Case Reports in Hematology
Mamiko Sakata-Yanagimoto, Shigeru Chiba
The molecular pathogenesis of peripheral T-cell lymphoma (PTCL) has gradually been clarified in terms of genomic abnormalities. Insights into these genomic abnormalities have provided clues to understanding the pathogenesis of PTCL. Furthermore, the origins of lymphoma cells have been clarified by investigating the distribution of genomic abnormalities in tumor cells and non-tumor blood cells. Multistep tumorigenesis has been suggested to be a fundamental mechanism underlying the development of angioimmunoblastic T-cell lymphoma (AITL), a distinct subtype of PTCL: premalignant cells evolve from hematopoietic progenitors via mutations in epigenetic regulators...
August 2016: [Rinshō Ketsueki] the Japanese Journal of Clinical Hematology
Ana E Suárez, M J Artiga, Carlos Santonja, Santiago Montes-Moreno, P De Pablo, Luis Requena, Miguel A Piris, Socorro M Rodríguez-Pinilla
Plasma cell proliferations in specific cutaneous lesions of angioimmunoblastic T-cell lymphoma(AITL) are very uncommon. Here, we report a case of clonal plasma cell proliferation in skin with heavy-chain-immunoglobulin-isotype-switch after cutaneous disease progression. Histopathologically, initial plaque lesions were suggestive of marginal-zone B-cell-lymphoma. Nevertheless, this 77-year-old lady was diagnosed with AITL after the progression of skin lesions from plaques to nodular tumors. A lymph node biopsy confirmed the diagnosis...
September 6, 2016: Journal of Cutaneous Pathology
Yi Zhou, Marc K Rosenblum, Ahmet Dogan, Achim A Jungbluth, April Chiu
Epstein-Barr virus (EBV)-associated B cell lymphoproliferative disorders may be seen in patients with angioimmunoblastic T cell lymphoma (AITL). Although both nodal and extranodal sites of involvement have been described, central nervous system involvement by B cell lymphoma following AITL has not previously been documented. We report a first example of such unusual presentation, in which an 80-year-old man developed diffuse large B cell lymphoma (DLBCL) in the cerebellum 4 months after the initial diagnosis of AITL...
December 2015: Journal of Hematopathology
Emilie Reboursiere, Fabien Le Bras, Charles Herbaux, Emmanuel Gyan, Aline Clavert, Franck Morschhauser, Sandra Malak, David Sibon, Florence Broussais, Thorsten Braun, Luc-Matthieu Fornecker, Reda Garidi, Sabine Tricot, Roch Houot, Bertrand Joly, Wajed Abarah, Bachra Choufi, Anne-Dominique Pham, Anne-Claire Gac, Christophe Fruchart, Emilie Marin, Violaine Safar, Anne Parcelier, Hervé Maisonneuve, Emmanuel Bachy, Guillaume Cartron, Arnaud Jaccard, Olivier Tournilhac, Cédric Rossi, Luciane Schirmer, Jean-Alain Martignoles, Philippe Gaulard, Hervé Tilly, Gandhi Damaj, Lymphoma Study Association Lysa Centers
Peripheral T-cell lymphoma (PTCL) is a group of diseases with poor outcome and few therapeutic options. We aimed to assess the efficacy of bendamustine in real life cohort of patients.Between November 2009 and March 2015, 138 PTCL patients were treated with bendamustine in 27 centers. Population median age was 64 (28-89) years with male/female ratio of 1.4. There were mainly angio-immunoblastic (AITL = 71), PTCL-not otherwise specified (PTCL-NOS = 40) and anaplastic large cell lymphoma (ALCL = 8). The majority of patients (96%) had disseminated disease and extranodal localizations (77%)...
July 21, 2016: Oncotarget
Daisuke Kurita, Hiroaki Miyoshi, Noriaki Yoshida, Yuya Sasaki, Seiichi Kato, Daisuke Niino, Yasuo Sugita, Yoshihiro Hatta, Masami Takei, Makoto Makishima, Koichi Ohshima
Lennert lymphoma (LeL) is a variant of peripheral T-cell lymphoma, not otherwise specified. Few clinicopathologic studies have investigated LeL, which is a rare disease. Here, we analyzed the clinicopathologic features of 26 patients with LeL to identify potential prognostic factors. Neoplastic cells positive for CD4, CD8, CD4/CD8, TIA-1, and granzyme B were observed in 21 (80.8%), 4 (15.4%), 1 (3.8%), 4 (15.3%), and 0 (0.0%) patients, respectively. Regarding follicular helper T-cell (TFH) markers, neoplastic cells positive for programmed cell death-1 (PD-1), CXCL13, CD10, and BCL6 were observed in 14 (53...
September 2016: American Journal of Surgical Pathology
David Vallois, Maria Pamela D Dobay, Ryan D Morin, François Lemonnier, Edoardo Missiaglia, Mélanie Juilland, Justyna Iwaszkiewicz, Virginie Fataccioli, Bettina Bisig, Annalisa Roberti, Jasleen Grewal, Julie Bruneau, Bettina Fabiani, Antoine Martin, Christophe Bonnet, Olivier Michielin, Jean-Philippe Jais, Martin Figeac, Olivier A Bernard, Mauro Delorenzi, Corinne Haioun, Olivier Tournilhac, Margot Thome, Randy D Gascoyne, Philippe Gaulard, Laurence de Leval
Angioimmunoblastic T-cell lymphoma (AITL) and other lymphomas derived from follicular T-helper cells (TFH) represent a large proportion of peripheral T-cell lymphomas (PTCLs) with poorly understood pathogenesis and unfavorable treatment results. We investigated a series of 85 patients with AITL (n = 72) or other TFH-derived PTCL (n = 13) by targeted deep sequencing of a gene panel enriched in T-cell receptor (TCR) signaling elements. RHOA mutations were identified in 51 of 85 cases (60%) consisting of the highly recurrent dominant negative G17V variant in most cases and a novel K18N in 3 cases, the latter showing activating properties in in vitro assays...
September 15, 2016: Blood
Colombe Saillard, Helene Guermouche, Coralie Derrieux, Julie Bruneau, Laurent Frenzel, Lucile Couronne, Vahid Asnafi, Elizabeth Macintyre, Amélie Trinquand, Ludovic Lhermitte, Thierry Molina, Felipe Suarez, Francois Lemonnier, Olivier Kosmider, Richard Delarue, Olivier Hermine, Morgane Cheminant
We report the case of a patient with a history of Epstein-Barr virus-positive large B-cell lymphoma, who relapsed with an angioimmunoblastic T-cell lymphoma (AITL) associated with a chronic myelomonocytic leukaemia (CMML). We performed targeted next-generation sequencing on CMML and AITL DNA, which revealed mutations of TET2, DNMT3A, SRSF2, NRAS and IDH1, thus confirming that the spectrum of AITL mutations share similarities with myeloid disorders. The frequencies of TET2/DNMT3A and SRSF2 variants could support the hypothesis that TET2/DNMT3A mutations occurred in an early progenitor cell, which later progressed to both the AITL and CMML clones...
June 29, 2016: Hematological Oncology
Harumi Kato, Kazuhito Yamamoto, Yusuke Higuchi, Hideyuki Yamamoto, Toko Saito, Hirofumi Taji, Yasushi Yatabe, Shigeo Nakamura, Tomohiro Kinoshita
There are few effective options for salvage therapy in elderly patients with relapsed or refractory angioimmunoblastic T-cell lymphoma (AITL). The anti-CCR4 antibody mogamulizumab works via antibody-dependent cytotoxic activity, reduces regulatory T cells, and evokes antitumor immunity in cancer patients. We report a 78-year-old patient with refractory AITL receiving a new immunochemotherapy consisting of sequential mogamulizumab administration followed by the GDP (gemcitabine, dexamethasone and cisplatin) regimen...
May 27, 2016: Chemotherapy
José R Cortés, Teresa Palomero
PURPOSE OF REVIEW: Once an obscure disease, recent studies have transformed our understanding of angioimmunoblastic T-cell lymphoma (AITL). In this review, we summarize new major advances in the genetics and biology of AITL. RECENT FINDINGS: Genome wide sequencing studies have dissected the repertoire of the genetic alterations driving AITL uncovering a highly recurrent Gly17Val somatic mutation in the small GTPase RHOA and major role for mutations in epigenetic regulators, such as TET2, DNMT3A and IDH2, and signaling factors (e...
July 2016: Current Opinion in Hematology
Ryoko Nagao, Yara Yukie Kikuti, Joaquim Carreras, Tomoki Kikuchi, Masashi Miyaoka, Hiromichi Matsushita, Minoru Kojima, Kiyoshi Ando, Mamiko Sakata-Yanagimoto, Shigeru Chiba, Naoya Nakamura
Angioimmunoblastic T-cell lymphoma (AITL) is an infrequent subtype of peripheral T-cell lymphoma derived from follicular helper T cells. Recently, a somatic G17V RHOA gene mutation has been reported. In this article, we examined the RHOA G17V mutation in 18 cases of AITL by 3 different techniques of Sanger sequencing, fully automated SNP genotyping, and deep sequencing, using routine diagnostic formalin-fixed paraffin-embedded tissue. The RHOA G17V mutation was detected in 10 cases (56%). Among the 10 mutated cases, 8 cases were detected by all 3 methods...
August 2016: American Journal of Surgical Pathology
Yuyue Ren, Yunhe Gu, Wei Wang, Jinghua Wang, Xiaoyun Li, Ying Wang, Haiyan Gao, Xiushuai Dong, Yaoyao Tian
Silent information regulator type-1 (SIRT1) is the best-studied member of the Sirtuin (Sir2) family of nicotinamide dinucleotide (NAD)-dependent class III histone deacetylases (HDACs). Rrecently, it is suggested that SIRT1 may be involved in the development of malignant tumors including mouse lymphoma, but has not yet been explored in Angioimmunoblastic T-cell lymphoma (AITL). Therefore, we investigated the prevalence and the prognostic impact of SIRT1 expression in AITL. Immunohistochemical expression of SIRT1, p53 were evaluated by using a 2 mm core from 45 AITL patients...
June 17, 2016: Cell Cycle
Sanam Loghavi, Sa A Wang, L Jeffrey Medeiros, Jeffrey L Jorgensen, Xin Li, Zijun Y Xu-Monette, Roberto N Miranda, Ken H Young
Angioimmunoblastic T-cell lymphoma (AITL) often shows systemic symptoms related to immune dysregulation and cytokine production. Biopsy usually harbors few malignant cells in an abundant reactive background, which can be diagnostically challenging in cases with small biopsies. This study was performed to assess the value of flow cytometry (FC) and to determine the immunophenotypic alterations in 155 samples from 38 patients with AITL. FC detected an aberrant T-cell population in 97 of 155 samples that represented 0...
April 22, 2016: Leukemia & Lymphoma
Hsiao-Wen Kao, Tung-Liang Lin, Lee-Yung Shih, Po Dunn, Ming-Chung Kuo, Yu-Shin Hung, Jin-Hou Wu, Tzung-Chih Tang, Hung Chang, Tseng-Tong Kuo, Che-Wei Ou, Po-Nan Wang
We retrospectively analyzed 87 patients with angioimmunoblastic T cell lymphoma (AITL) in Taiwan. The median age was 68 (range 18-89) years. Of these patients, 74 % was at an advanced stage. The most common extra-nodal site involved was bone marrow (36 %). Of these patients, 77 % were International Prognostic Index (IPI) >1 and 79 % had a prognostic index for peripheral T-cell lymphoma (PIT) >1. Of 75 patients who received systemic chemotherapy, the complete remission rate was 60 %, the relapse rate was 47 %, and the 2-year progression-free survival rate was 37...
August 2016: International Journal of Hematology
Hua-Ping DU, Qian-Qian Yang, Y E Zhang
The peripheral T-cell lymphomas (PTCLs) are a heterogeneous group of aggressive neoplasms that account for <15% of all non-Hodgkin's lymphoma cases in adults. Angioimmunoblastic T-cell lymphoma (AITL) is a specific subtype of PTCL. The tumor is frequently aggressive and there is currently no general consensus regarding an effective treatment strategy. The present study reports a case in which bortezomib combined with dexamethasone was used to treat refractory AITL. A 63-year-old woman was admitted to Sir Run Run Shaw Hospital, Zhejiang University School of Medicine (Zhejiang, China) on August 17, 2013...
March 2016: Oncology Letters
Yoshimi Ishii, Megumi Itabashi, Ayumi Numata, Wataru Yamamoto, Kenji Motohashi, Maki Hagihara, Kenji Matsumoto, Shin Fujisawa
Angioimmunoblastic T-cell lymphoma (AITL) is an aggressive T-cell lymphoma. A 63-year-old man was diagnosed with AITL. He received 6 cycles of CHOP therapy, but showed progressive disease. Subsequently, he received ESHAP chemotherapy; however, it was not effective. He received mogamulizumab (an anti-CCR4 monoclonal antibody). After 4 cycles, his respiratory condition worsened and he was diagnosed with cytomegalovirus (CMV) pneumonia. Despite antiviral and antibiotic therapy, he died. We speculate that the combination of progressive lymphoma with mogamulizumab and chemotherapy likely caused CMV pneumonia...
2016: Internal Medicine
Scott V Adams, Polly A Newcomb, Andrei R Shustov
PURPOSE: To compare incidence and survival of peripheral T-cell lymphoma (PTCL) subtypes among US racial/ethnic groups. METHODS: Patients with PTCL (age ≥ 15 years; 2000 to 2012) were identified in the Surveillance, Epidemiology, and End Results (SEER) registries. Race/ethnicity was categorized as non-Hispanic white, black, Asian/Pacific Islander, Hispanic white, or American Indian/Alaskan native. Age-standardized annual incidence rates and incidence rate ratios were estimated with 95% CIs, and case-case odds ratios were estimated by race/ethnicity using polytomous regression...
March 20, 2016: Journal of Clinical Oncology: Official Journal of the American Society of Clinical Oncology
Mohleen Kang, Nitasha Bhatia, Adrienne Sauder, Mirela Feurdean
Angioimmunoblastic T cell lymphoma (AITL) is a rare but distinct type of T cell lymphoma with an aggressive course and high mortality. Most patients are diagnosed late in the disease and usually present with generalized lymphadenopathy. A minority have skin lesions at the time of diagnosis, more commonly in the form of nonspecific maculopapular rash with or without pruritus. We report a rare case of AITL presenting with chronic, recurrent angioedema and urticaria-like lesions and no palpable peripheral adenopathy...
2016: Case Reports in Medicine
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