Multifactorial neutropenia in a patient with acute promyelocytic leukemia and associated large granular lymphocyte expansion: A case report

Reda, Gianluigi; Fattizzo, Bruno; Cassin, Ramona; Flospergher, Elena; Orofino, Nicola; Gianelli, Umberto; Barcellini, Wilma; Cortelezzi, Agostino

doi:10.3892/ol.2016.5549

Cited by 6 publications

(6 citation statements)

References 10 publications

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“…A case of steroid-responsive AIHA has been reported in post-MDS acute myeloid leukemia (AML) [ 56 ]. Finally, AIN and steroid-refractory ITP has been documented in patients with acute promyelocytic leukemia [ 61 , 62 ].…”

Section: Autoimmune Complications In Myeloid Neoplasmsmentioning

confidence: 99%

Autoimmune Complications in Hematologic Neoplasms

Barcellini

Giannotta

Fattizzo

2021

Cancers

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Autoimmune cytopenias (AICy) and autoimmune diseases (AID) can complicate both lymphoid and myeloid neoplasms, and often represent a diagnostic and therapeutic challenge. While autoimmune hemolytic anemia (AIHA) and immune thrombocytopenia (ITP) are well known, other rarer AICy (autoimmune neutropenia, aplastic anemia, and pure red cell aplasia) and AID (systemic lupus erythematosus, rheumatoid arthritis, vasculitis, thyroiditis, and others) are poorly recognized. This review analyses the available literature of the last 30 years regarding the occurrence of AICy/AID in different onco-hematologic conditions. The latter include chronic lymphocytic leukemia (CLL), lymphomas, multiple myeloma, myelodysplastic syndromes (MDS), chronic myelomonocytic leukemia (CMML), myeloproliferative neoplasms, and acute leukemias. On the whole, AICy are observed in up to 10% of CLL and with higher frequencies in certain subtypes of non-Hodgkin lymphoma, whilst they occur in less than 1% of low-risk MDS and CMML. AID are described in up to 30% of myeloid and lymphoid patients, including immune-mediated hemostatic disorders (acquired hemophilia, thrombotic thrombocytopenic purpura, and anti-phospholipid syndrome) that may be severe and fatal. Additionally, AICy/AID are found in about 10% of patients receiving hematopoietic stem cell transplant or treatment with new checkpoint inhibitors. Besides the diagnostic difficulties, these AICy/AID may complicate the clinical management of already immunocompromised patients.

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Section: Autoimmune Complications In Myeloid Neoplasmsmentioning

confidence: 99%

Autoimmune Complications in Hematologic Neoplasms

Barcellini

Giannotta

Fattizzo

2021

Cancers

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“…Moreover, these mechanisms are not mutually exclusive but interact with each other in a patient with T-LGL [16]. Recently, a case report of an acute promyelocytic leukemia patient with chronic severe neutropenia and related LGL expansion also suggested the multifactorial origin of neutropenia and that immune activation/dysregulation may be conducive to the progression of neutropenia [18].…”

Section: Abnormal Neutropenia In Lgl Leukemia Patientsmentioning

confidence: 99%

Dysfunction of immune system in the development of large granular lymphocyte leukemia

2018

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Objectives: Large granular lymphocyte (LGL) leukemia is a rare type of lymphoproliferative disease caused by clonal antigenic stimulation of T cells and natural killer (NK) cells. Methods: In this review, we focus on the current knowledge of the immunological dysfunctions associated with LGL leukemia and the associated disorders coexistent with this disease. Novel therapeutic options targeting known molecular mechanisms are also discussed. Results and Discussion: The pathogenesis of LGL leukemia involves the accumulation of gene mutations, dysregulated signaling pathways and immunological dysfunction. Mounting evidence indicated that dysregulated survival signaling pathways may be responsible for the immunological dysfunction in LGL leukemia including decreased numbers of neutrophils, dysregulated signal transduction of NK cells, abnormal B-cells, aberrant CD8+ T cells, as well as autoimmune and hematological abnormalities. Conclusion: A better understanding of the immune dysregulation triggered by LGL leukemia will be beneficial to explore the pathogenesis and potential therapeutic targets for this disease.

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“…LGL clones have been also described in myeloproliferative neoplasms (MPN) and AML, even though this association is rarer (Table 2). There are case reports describing the association of LGL clones with AML, acute promyelocytic leukemia (APL), essential thrombocythemia, chronic myeloid leukemia (CML), and chronic myelomonocytic leukemia (38)(39)(40)(41)(42)(43)(44). Again, a hallmark of this association is cytopenia.…”

Section: Lgl Expansion In Other Myeloid Neoplasmsmentioning

confidence: 99%

“…Despite chemotherapy, LGLL clone continued to increase, possibly due to a growing advantage after leukemic depletion. Interestingly, severe neutropenia persisted and also interfered with chemotherapy maintenance (44). Finally, even for LGL/AML, it has been speculated that the two clones may origin from the same progenitor (38) and this association did not lead to a worse prognosis.…”

Section: Lgl Expansion In Other Myeloid Neoplasmsmentioning

confidence: 99%

Large Granular Lymphocyte Expansion in Myeloid Diseases and Bone Marrow Failure Syndromes: Whoever Seeks Finds

et al. 2021

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Large granular lymphocytes (LGL) are lymphoid cells characterized by either a T-cell or a natural killer phenotype whose expansion may be reactive to toxic, infectious, and neoplastic conditions, or result from clonal selection. Recently, the higher attention to LGL clones led to their detection in many clinical conditions including myeloid neoplasms and bone marrow failures. In these contexts, it is still unclear whether LGL cells actively contribute to anti-stem cell autoimmunity or are only a reaction to dysplastic/leukemic myelopoiesis. Moreover, some evidence exists about a common clonal origin of LGL and myeloid clones, including the detection of STAT3 mutations, typical of LGL, in myeloid precursors from myelodysplastic patients. In this article we reviewed available literature regarding the association of LGL clones with myeloid neoplasms (myelodysplastic syndromes, myeloproliferative neoplasms, and acute myeloid leukemias) and bone marrow failures (aplastic anemia and pure red cell aplasia, PRCA) focusing on evidence of pathogenic, clinical, and prognostic relevance. It emerged that LGL clones may be found in up to one third of patients, particularly those with PRCA, and are associated with a more cytopenic phenotype and good response to immunosuppression. Pathogenically, LGL clones seem to expand after myeloid therapies, whilst immunosuppression leading to LGL depletion may favor leukemic escape and thus requires caution.

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Multifactorial neutropenia in a patient with acute promyelocytic leukemia and associated large granular lymphocyte expansion: A case report

Cited by 6 publications

References 10 publications

Autoimmune Complications in Hematologic Neoplasms

Autoimmune Complications in Hematologic Neoplasms

Dysfunction of immune system in the development of large granular lymphocyte leukemia

Large Granular Lymphocyte Expansion in Myeloid Diseases and Bone Marrow Failure Syndromes: Whoever Seeks Finds

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