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LETTER TO THE EDITOR
Year : 2015  |  Volume : 52  |  Issue : 2  |  Page : 193-194
 

T-cell chronic lymphocytic leukemia/T-prolymphocytic leukemia: Experience with single agent bendamustine


1 Department of Medical Oncology, Nizam's Institute of Medical Sciences, Hyderabad, Andhra Pradesh, India
2 Department of Pathology, Nizam's Institute of Medical Sciences, Hyderabad, Andhra Pradesh, India

Date of Web Publication5-Feb-2016

Correspondence Address:
Gundeti Sadashivudu
Department of Medical Oncology, Nizam's Institute of Medical Sciences, Hyderabad, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-509X.175812

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How to cite this article:
Thota N K, Sadashivudu G, Gandhi LV, Pragnya C, Roshni T P, Raghunadharao D. T-cell chronic lymphocytic leukemia/T-prolymphocytic leukemia: Experience with single agent bendamustine. Indian J Cancer 2015;52:193-4

How to cite this URL:
Thota N K, Sadashivudu G, Gandhi LV, Pragnya C, Roshni T P, Raghunadharao D. T-cell chronic lymphocytic leukemia/T-prolymphocytic leukemia: Experience with single agent bendamustine. Indian J Cancer [serial online] 2015 [cited 2021 Aug 5];52:193-4. Available from: https://www.indianjcancer.com/text.asp?2015/52/2/193/175812


Sir,

T-cell chronic lymphocytic leukemia is now reclassified as T-prolymphocytic leukemia (T-PLL) according to the World Health Organization/Revised European-American Classification of lymphoid neoplasm's and it represents approximately 30% of T-cell leukemia's with a mature cell phenotype. Presentation usually in the sixth decade or later and occurs more frequently in males. Characteristically, T-PLL presents with rapidly increasing lymphocyte count, splenomegaly, low volume lymphadenopathy skin rashes, peripheral edema, and pleuroperitoneal effusion.

The optimal treatment for this disease in not known. Several case reports and case series showed the response rates ranging from 25% to 45% to purine analogs, CHOP, (cyclophosphamide, doxorubicin, vincristine and prednisolone) mitoxantrone and cytarabine with a median survival of approximately 1 year.[1] Results with alemtuzumab, an anti-CD52 monoclonal antibody in relapsed and refractory T-PLL showed an overall response rate (ORR) and complete remission rate of 76% with 60% respectively, with a median overall survival (OS) of 16 months in those who obtained a complete remission.[2] Hopfinger et al.,[3] published results of an induction regimen of fludarabine, mitoxantrone and cyclophosphamide followed by alemtuzumab consolidation in treatment naive T-PLL patients. The ORR was 68% with a median OS of 17.1 months. Median survival of 48 months was observed with alemtuzumab followed by the stem cell transplant.[4] We present the first case report of treatment with bendamustine in a patient with T-PLL.

A 37-year-old gentleman presented with 2 months history of swellings in the neck, armpits and groins with un-quantified weight loss. Examination revealed generalized lymphadenopathy and mild splenomegaly. The hemogram showed a hemoglobin of 13.0 g/dL, white blood count of 1,67,000/mm, platelets count of 210 × 109/L with 81% of small mature lymphocytes, 5% of prolymphocytes, 10% of neutrophils, 4% of eosinophils and few smudge cells. Bone marrow aspiration and biopsy were particulate with an increased cellularity and prominence of small mature lymphocytes accounting for 89% of the cells. Immunohistochemistry on bone marrow biopsy was positive for T-cell marker CD3 and negative for B cell markers. Flow cytometry on the marrow aspirate revealed abnormal lymphocytes, which were positive for T-cell markers CD2, CD3, CD4, CD5, CD 8, and negative for B Cell markers CD 19, CD 20, CD 22, sIgM, Kappa, Lambda, CD19 + ZAP, CD19, CD5 [Figure 1]. A diagnosis of T-PLL was made.
Figure 1: Flow cytometry on bone marrow aspirate showing positivity for T-cell markers

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After explaining the treatment options, written informed consent was taken and he was started on a single agent bendamustine. He received 6 cycles of chemotherapy every 4 weeks at a dose of 100 mg/m 2 on day 1 and day 2. He responded clinically and his hemogram normalized at the end of the second cycle. He tolerated chemotherapy very well without any dose compromise and major toxicity. End-of-therapy bone marrow evaluation showed complete remission and flow cytometry for minimal residual disease was less than 0.1% [Figure 2]. He is on follow-up for the last 36 months without progression.
Figure 2: Flow cytometry on bone marrow aspirate, at the end of therapy, showing minimal residual disease

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This case suggests that bendamustine either as a single agent or in combination for the management of T-PLL deserves further study.

 
  References Top

1.
Matutes E, Brito-Babapulle V, Swansbury J, Ellis J, Morilla R, Dearden C, et al. Clinical and laboratory features of 78 cases of T-prolymphocytic leukemia. Blood 1991;78:3269-74.  Back to cited text no. 1
    
2.
Dearden CE, Matutes E, Cazin B, Tjønnfjord GE, Parreira A, Nomdedeu B, et al. High remission rate in T-cell prolymphocytic leukemia with CAMPATH-1H. Blood 2001;98:1721-6.  Back to cited text no. 2
    
3.
Hopfinger G, Busch R, Pflug N, Weit N, Westermann A, Fink AM, Cramer P, et al. Sequential chemoimmunotherapy of fludarabine, mitoxantrone, and cyclophosphamide induction followed by alemtuzumab consolidation is effective in T-cell prolymphocytic leukemia. Cancer 2013;119:2258-67.  Back to cited text no. 3
    
4.
Krishnan B, Else M, Tjonnfjord GE, Cazin B, Carney D, Carter J, et al. Stem cell transplantation after alemtuzumab in T-cell prolymphocytic leukaemia results in longer survival than after alemtuzumab alone: A multicentre retrospective study. Br J Haematol 2010;149:907-10.  Back to cited text no. 4
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