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Published on: February 21, 2018
Granular lymphocytic leukemia derived from gamma delta T-cell expressing cytotoxic molecules
T Saito1, K Togitani, J Murakami
1Hematology Division, National Cancer Center Hospital, 5-1-1, Tsukiji, Chuo-ku, Tokyo 104-0045, Japan.
Insights
This report details an exceptionally rare case of granular lymphocytic leukemia originating from gamma delta T-cells (gamma delta T-GLL). This finding highlights a unique subtype of leukemia with distinct immunophenotypic characteristics.
Area of Science:
- Hematology
- Immunology
- Oncology
Background:
- Granular lymphocytic leukemia (GLL) is a rare hematologic malignancy.
- Gamma delta (γδ) T-cells represent a distinct lineage of T lymphocytes with unique functions.
- The occurrence of GLL specifically derived from γδ T-cells is exceptionally uncommon.
Observation:
- A patient presented with leukocytosis (25.7 x 10^9/L) comprising 94% atypical lymphocytes with cytoplasmic granules.
- Immunophenotyping revealed lymphocytes positive for CD2, CD3, CD5, CD7, CD56, and T-cell receptor (TCR) γδ, but negative for CD4, CD8, CD57, TCR αβ, and B-cell antigens.
- Cytotoxic molecules T-cell intracellular antigen-1 (TIA-1) and granzyme B were positive, indicating cytotoxic potential.
Findings:
- Southern blot analysis confirmed T-cell receptor J gamma and C beta gene rearrangement, with germline configuration of the JH gene.
- Human T-cell leukemia virus type-I (HTLV-I) infection was ruled out through serological and molecular testing.
- Imaging revealed splenomegaly without lymphadenopathy.
Implications:
- The case establishes a diagnosis of gamma delta T-cell granular lymphocytic leukemia (γδ T-GLL).
- The patient was managed conservatively with over 4 years of follow-up without therapeutic intervention.
- This case contributes to the understanding of rare T-cell malignancies and the spectrum of γδ T-cell neoplasms.
Abstract:
We here present an extremely rare case of granular lymphocytic leukemia derived from gamma delta T-cell (gamma delta T-GLL). The blood picture at diagnosis was as follows; white cell count 25.7 x 10(9)/l containing 94% atypical lymphocytes with cytoplasmic granules, hemoglobin 11.8 g/dl and platelet count 124 x 10(9)/l. The atypical lymphocytes were positive for CD2, CD3, CD5, CD7, CD56 and TCR gamma delta, but negative for CD4, CD8, CD57, TCR alpha beta and B-cell antigens. The cytotoxic molecules, T-cell intracellular antigen-1 (TIA-1) and granzyme B, were positive by immunocytochemical analysis. Southern blot analysis showed rearrangement of T-cell receptor J gamma and C beta genes but germline configuration of the JH gene. Neither serum antibody against human T-cell leukemia virus type-I (HTLV-I) nor the integration of HTLV-I proviral DNA was detected. CT scan showed splenomegaly but no lymph node enlargement. A diagnosis of gamma delta T-GLL was made, and she has been followed up without any therapies for more than 4 years.
