Mediastinal T-cell lymphoma with presumed secondary monostotic long bone involvement in the absence of local recurrence

2020 ◽  
Vol 8 (1) ◽  
pp. e000904
Author(s):  
Andrea Mosca ◽  
Oliver Greville-Heygate ◽  
Fernando Constantino-Casas ◽  
Antonio Giuliano ◽  
Jane M Dobson

A 7-year-old male neutered cocker crossbreed was presented with a 1-month history of tachypnoea and exercise intolerance. Initial investigations confirmed a pleural effusion due to a mediastinal T-cell lymphoma. L-asparaginase and a 23-week lomustine, vincristine, prednisolone protocol were pursued and complete response was recorded at the end of the protocol. Ten months later, he was presented at the referring veterinary practice with a non-bearing lameness of his left forelimb, and a transcondylar screw was placed following the suspicion of a fracture of the lateral humeral epicondyle. During surgery, a sample of bone was collected and submitted for histopathology. Histopathology and immunohistochemistry were consistent with a T-cell lymphoma. Total body CT scan revealed a monostotic aggressive lesion of the distal humerus with no evidence of infiltration into bone at any other sites, and no other signs of lymphoma relapse were seen elsewhere in the body. Based on these findings, secondary bone lymphoma was hypothesised.

2021 ◽  
Vol 17 (1) ◽  
Author(s):  
Sara Kotb ◽  
Carolina Allende ◽  
T. William O’Neill ◽  
Krista Bruckner ◽  
Helio DeMorais ◽  
...  

Abstract Background Renal lymphoma in dogs is rare and has a poor prognosis. Granular lymphocyte morphology is rarely reported in canine renal lymphoma. Mild to moderate polycythemia is reported in a number of canine renal lymphoma cases. Case presentation A 10-year-old Labrador retriever presented to a university veterinary teaching hospital after a 1-month history of polyuria, polydipsia, and pollakiuria and a 2-week history of abdominal distention, lethargy, and increased respiratory effort. Abdominal ultrasound showed a wedge-shaped to rounded, heterogeneously hypoechoic mass lesion in the left kidney. Cytologic analysis of a percutaneous aspirate of the mass was consistent with lymphoma of granular lymphocytes. Severe polycythemia (hematocrit 0.871) was noted on a complete blood cell count. Clonality analysis identified a clonally rearranged T-cell receptor (TCR) gene and immunohistochemical staining was CD3+, CD79a- and CD11d+, supporting cytotoxic T-cell lymphoma. Conclusions To our knowledge, this is the first report of renal cytotoxic T-cell lymphoma with severe polycythemia in a dog. Severe polycythemia and renal cytotoxic T-cell lymphoma are both rare in dogs; this report adds to the body of knowledge on these conditions.


2012 ◽  
Vol 30 (6) ◽  
pp. 631-636 ◽  
Author(s):  
Bertrand Coiffier ◽  
Barbara Pro ◽  
H. Miles Prince ◽  
Francine Foss ◽  
Lubomir Sokol ◽  
...  

Purpose Romidepsin is a structurally unique, potent class 1 selective histone deacetylase inhibitor. The primary objective of this international, pivotal, single-arm, phase II trial was to confirm the efficacy of romidepsin in patients with relapsed or refractory peripheral T-cell lymphoma (PTCL). Patients and Methods Patients who were refractory to at least one prior systemic therapy or for whom at least one prior systemic therapy failed received romidepsin at 14 mg/m2 as a 4-hour intravenous infusion on days 1, 8, and 15 every 28 days. The primary end point was the rate of complete response/unconfirmed complete response (CR/CRu) as assessed by an independent review committee. Results Of the 131 patients enrolled, 130 had histologically confirmed PTCL by central review. The median number of prior systemic therapies was two (range, one to eight). The objective response rate was 25% (33 of 130), including 15% (19 of 130) with CR/CRu. Patient characteristics, prior stem-cell transplantation, number or type of prior therapies, or response to last prior therapy did not have an impact on response rate. The median duration of response was 17 months, with the longest response ongoing at 34+ months. Of the 19 patients who achieved CR/CRu, 17 (89%) had not experienced disease progression at a median follow-up of 13.4 months. The most common grade ≥ 3 adverse events were thrombocytopenia (24%), neutropenia (20%), and infections (all types, 19%). Conclusion Single-agent romidepsin induced complete and durable responses with manageable toxicity in patients with relapsed or refractory PTCL across all major PTCL subtypes, regardless of the number or type of prior therapies. Results led to US Food and Drug Administration approval of romidepsin in this indication.


2007 ◽  
Vol 135 (1-2) ◽  
pp. 80-84
Author(s):  
Milena Bakrac ◽  
Branka Bonaci-Nikolic ◽  
Natasa Colovic ◽  
Sanja Simic-Ogrizovic ◽  
Miodrag Krstic ◽  
...  

Enteropathy associated T-cell lymphoma (EATCL) is a high grade, pleomorphic peripheral T-cell lymphoma with usually cytotoxic phenotype. This is a case report of three patients with EATCL. The first patient was 50 year-old woman with four year history of gluten sensitive enteropathy (GSE). Diagnosis of lymphoma was confirmed after the resection of the jejunum (small intestine obstruction). Pathohistological (PAS, Reticulin, Giemsa) and immunohistochemical (anti-LCA, anti-CD20, anti- CD45RO, anti-CD3) methods revealed the diagnosis of EATCL: CD45RO+, CD3+. After the third cycle of chemotherapy, the disease progressed with massive lung infiltration. Patient died due to complications of bone marrow aplasia. The second patient was 23 year-old woman with long earlier history of GSE. She presented with the acute renal failure. According to established diagnosis of tubulointerstitial nephritis, she was treated with pulse doses of steroid therapy. After temporary improvement, she had dissemination of the disease. On MRI, small intestinal wall was thickened, and abdominal lymph nodes were enlarged with extraluminal compression of common bile duct. Laparotomy with mesenterial lymph node biopsy and consecutive pathohistological and immunohistochemical analyses revealed the diagnosis of EATCL. The patient received chemotherapy, but she died with signs of pulmonary embolization. The third patient was 53 year-old woman without previous history of GSE. Diagnosis of EATCL was revealed after the resection of jejunum because of small intestinal obstruction. She received two cycles of chemotherapy, but she died with signs of disease progression. IgA antiendomysial antibodies were detected in the serum of all patients. The overall survival of patients was 7 months. The possibility of lymphoma rising in patients with clinical progression of GSE despite gluten free diet must be kept in mind.


2021 ◽  
Vol 11 ◽  
Author(s):  
Laura Ballotta ◽  
Pier Luigi Zinzani ◽  
Stefano Pileri ◽  
Riccardo Bruna ◽  
Monica Tani ◽  
...  

Patients with relapsed/refractory (R/R) peripheral T-cell lymphoma (PTCL) have a poor prognosis, with an expected survival of less than 1 year using standard salvage therapies. Recent advances in our understanding of the biology of PTCL have led to identifying B-Cell Lymphoma 2 (BCL2) protein as a potential therapeutic target. BLC2 inhibitor venetoclax was investigated in a prospective phase II trial in patients with BCL2-positive R/R PTCL after at least one previous standard line of treatment (NCT03552692). Venetoclax given alone at a dosage of 800 mg/day resulted in one complete response (CR) and two stable diseases (SDs) among 17 enrolled patients. The majority of patients (88.2%) interrupted the treatment due to disease progression. No relationship with BCL2 expression was documented. At a median follow-up of 8 months, two patients are currently still on treatment (one CR and one SD). No case of tumor lysis syndrome was registered. Therefore, venetoclax monotherapy shows activity in a minority of patients whose biological characteristics have not yet been identified.Clinical Trial Registrationwww.clinicaltrials.gov (NCT03552692, EudraCT number 2017-004630-29).


1987 ◽  
Vol 5 (5) ◽  
pp. 750-755 ◽  
Author(s):  
R Liang ◽  
D Todd ◽  
T K Chan ◽  
K L Wong ◽  
F Ho ◽  
...  

Thirty-one Chinese patients with peripheral T cell lymphoma (PTCL) were reviewed. Using the modified Japanese Lymphoma Group classification, there were nine (29%) of the pleomorphic type, 16 (52%) immunoblastic lymphadenopathy (IBL)-like, two (7%) T-zone lymphoma, and one (3%) Lennert's lymphoepithelioid type. Three (9%) were not classifiable. All were positive for T11 (E rosette receptor antigen). Fifty-four percent (15 of 28) were positive predominantly for T4 (helper T cell) and 46% (13/28) for T8 (suppressor T cell). The median age of the patients was 57 years. They usually presented with advanced disease, and while extranodal involvement was common, CNS disease was not seen. The IBL-like type was associated with a positive Coombs' test and polyclonal hypergammaglobulinemia. Five of the nine pleomorphic type were checked for antibody to HTLV-I virus and all were negative. PTCL was associated with poor prognosis, which was not influenced by the histologic subtypes and the T4/T8 phenotypes. The complete response rate of 13 consecutive patients who received the BACOP (bleomycin, doxorubicin, cyclophosphamide, vincristine, and prednisone) L17M regimen was significantly better than the 16 historic controls who received other less-intensive regiments, 84% v 19% (P less than .01). The relapse rate was also significantly lower, 9% v 100% (P less than .001). There appeared to be an improvement in the disease-free survival (DFS) (80% v 0% at 18 months), as well as the overall survival (60% v 36% at 18 months), but the differences did not reach statistical significance due to small sample sizes.


2020 ◽  
pp. 107815522096861
Author(s):  
Lucie Oberic ◽  
Faustine Delzor ◽  
Caroline Protin ◽  
Sophie Perriat ◽  
Camille Laurent ◽  
...  

Introduction Brentuximab vedotin (Bv) has been approved for the treatment of Refractory/Relapsed (R/R) Anaplastic Large Cell Lymphomas (ALCL) and cutaneous T-Cell Lymphomas, but is also effective in other CD30+ malignancies. We report here the outcomes of patients with various R/R Peripheral T Cell Lymphoma (PTCL) treated with Bv in real life practice. Method This was a retrospective, single-center study based on medical records of patients with R/R PTCL treated either with Bv alone or in combination with chemotherapy. Results Among 27 patients treated with Bv, neutropenia was the main serious adverse event observed in particular when Bv was used as combination treatment. The complete Response Rates (CRR) was 40.7%; it was significantly improved when Bv was used as combination treatment. The majority of eligible patients (7/10) underwent Stem Cell Transplantation. Median Progression Free Survival (PFS) and Overall Survival (OS) were 5.2 months and 12.5 months respectively. Conclusion Our current study shows that Bv used in combination with chemotherapy provides a high CRR and thereby allows SCT in R/R PTCL. The use of Bv treatments in this setting warrants further investigation.


2016 ◽  
Vol 20 (3) ◽  
pp. 244-248 ◽  
Author(s):  
Catherine Besner Morin ◽  
David Roberge ◽  
Irina Turchin ◽  
Tina Petrogiannis-Haliotis ◽  
Gizelle Popradi ◽  
...  

Background: Numerous treatments are available for cutaneous T-cell lymphoma (CTCL), including systemic retinoids. Very few data are available on topical retinoids. Objectives: The aim of this study was to evaluate the safety and efficiency of tazarotene as monotherapy for early-stage CTCL. Methods: An open-label, prospective study of tazarotene as monotherapy for stages IA to IIA CTCL was conducted. Index lesions on 10 patients were followed for 6 months on treatment, plus at least 6 months off treatment. Results: Six patients (60%) showed complete response (CR). Erythema, scaling, thickness, and lesion area decreased progressively throughout treatment. The mean time to CR was 3.8 months; CR was durable for at least 6 months in 83%. Of the 4 patients (40%) without CR, 2 (20%) had stable disease and 2 (20%) stopped the medication because of local side effects; none showed progression. Conclusions: This is the first Canadian trial providing evidence that topical tazarotene has excellent potential as a monotherapy agent for stages I to IIA CTCL.


Blood ◽  
2009 ◽  
Vol 114 (22) ◽  
pp. 2939-2939
Author(s):  
Dae Ro Choi ◽  
Dok Hyun Yoon ◽  
Heui June Ahn ◽  
Yoojin Cho ◽  
Eun Kyoung Kim ◽  
...  

Abstract Abstract 2939 Poster Board II-915 Introduction: Peripheral blood absolute lymphocyte count (ALC) at the time of diagnosis is a prognostic indicator in hematologic malignancies. However, no reports have addressed whether ALC at the time of first relapse (ALC-R) has a prognostic significance in the patients with relapsed T-cell Non-Hodgkin's lymphoma (NHL). We retrospectively studied the prognostic significance of ALC-R in these patients. Patients and Methods: We identified 63 patients who had a documented relapsed disease after having reached a complete response, including at least an unconfirmed complete response between 1993 and 2007 and we analyzed their ALC-R and following variables at the time of first relapse; age, gender, the number of extranodal sites, lactate dehydrogenase, ECOG performance status, stage and international prognostic index. Results: Out of the 63 patients, 23 (36.5%) had a peripheral T-cell lymphoma, not otherwise characterized, while 15 (23.8%) had an extranodal NK/T-cell lymphoma, nasal type, 6 (9.5%) anaplastic large-cell lymphoma, 5 (7.9%) angioimmunoblastic T-cell lymphoma, 2 (3.2%) primary cutaneous T cell lymphoma and 12 (19%) other types. Among them, 32 (50.8%) had an ALC-R ≥ 1.25 × 109/L, 47 (74.6%) had an ECOG PS 0 or 1 and by IPI at relapse, 0, 1, 2, 3, 4, and 5 were 20.6%, 25.4%, 23.8%, 23.8%, 4.8%, and 1.6%, respectively. Univariate analyses showed that good ECOG PS (HR, 0.408; 95% CI 0.190-0.876; p=0.022) and high ALC at relapse (HR, 0.394; 95% CI 0.210-0.740; p=0.004) were associated with longer survival from relapse (Table 1). Multivariate analysis also showed that high ALC at relapse (HR, 0.369; 95% CI 0.187-0.726; p=0.004) and good ECOG PS (HR, 0.295; 95% CI 0.131-0.666; p=0.003) were still associated with longer survival outcome (Table 2). Conclusions: The high ALC-R predicted a better prognosis in patients with relapsed T-cell NHL, suggesting that the host immune system might have a crucial role. Disclosures: No relevant conflicts of interest to declare.


Blood ◽  
2009 ◽  
Vol 114 (22) ◽  
pp. 2730-2730
Author(s):  
Jennifer E. Amengual ◽  
Bruce G. Raphael

Abstract 2730 Poster Board II-706 Angioimmunoblastic T-cell lymphoma (AITL) is a rare subtype representing 2% of Non-Hodgkin's Lymphoma characterized by lymphadenopathy, hepatosplenomegaly, anemia, hypergammaglobulinemia and immune dysfunction. Prognosis is poor with a median survival of less than 36 months. There is no standard treatment for AITL. Most patients initially respond to treatment, but relapse within short time intervals. Alemtuzumab is a humanized monoclonal antibody that binds to CD52 antigen, a cell surface glycoprotein with high expression on T-cells. We report three patients with refractory AITL, with confirmed T-cell receptor gene rearrangements, who achieved sustained, durable responses with alemtuzumab. The table below lists the treatment regimens, duration of remissions and complications for all 3 cases. Infectious and autoimmune complications were effectively treated in all.PatientPrevious Treatment (Response duration, months)Alemtuzumab (Response duration)InfectionsAutoimmune manifestationsACHOP (10) Cytoxan-P (1) Gemzar-P (1)24 monthsCMVBCHOPE (10) ICE (0) Gemcitabine-P followed by Cyclosporine maintenance (1)>24 monthsAspergillusAgranulocytosis Autoimmune hemolytic anemiaCCHOP (1)>14 monthsCMV Legionella Patient A was a 73 year-old female who presented with lymphadenopathy and biopsy proven AITL. Her longest remission was 10 months following CHOP. She was started on alemtuzumab 30 mg 3 times per week for 4 weeks in June 2007 after relapsing. Her only complication from treatment was CMV infection. She remained in remission until June 2009 when she relapsed in her liver and colon. She was treated with alemtuzumab and prednisone for 2 weeks, but developed neutropenic fever, CMV and died July 2009. Patient B is a 73 year-old male with a history of ITP who presented in July 2005 with fevers, lymphadenopathy and anemia, and biopsy proven AITL. His longest remission was 10 months with CHOPE. In June 2007, the patient was treated with alemtuzumab for 7 weeks after relapsing. Treatment complications included Aspergillus pneumonia, agranulocytosis and autoimmune hemolytic anemia. He achieved a complete response as evidenced by PET/CT scan. He remains in remission 2 years later. Patient C is a 62 year-old woman with a history of MGUS who presented in 2007 with rapidly growing lymphadenopathy and a biopsy that revealed AITL. She never achieved a sustained remission with chemotherapy. June 2008, the patient was treated with alemtuzumab for 6 weeks, complicated by CMV and Legionella pneumonia. She remains in remission now over 14 months. Here we have shown remarkable success with short courses of alemtuzumab. Three patients remained disease free for an average of 21 months; two remissions are on-going. This report demonstrates sustained responses for patients with AITL, suggesting that alemtuzumab is a valid and rational treatment choice in heavily pretreated patients. We propose using anti-CD52 therapy as consolidation after primary response to conventional chemotherapy in patients with AITL. Disclosures: Off Label Use: Alemtuzumab is not licensed for use in Angioimmunoblastic T-cell Lymphoma.


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