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      Manifestación oral de linfoma linfoblástico de células-T: un caso inusual y desafiante Translated title: Oral manifestation of T-cell lymphoblastic lymphoma: an unusual and challenging case

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          Abstract

          RESUMEN Las neoplasias malignas de origen linfoide corresponden a un complejo grupo de enfermedades cuyo diagnóstico es complicado, ya que requiere la aplicación de pruebas inmunohistoquímicas, un exhaustivo análisis por parte del equipo histopatológico, así como también una muestra representativa del tejido a examinar. Se presenta el caso de un paciente previamente diagnosticado con linfoma no-Hodgkin estirpe T, con manifestación clínica en cavidad oral, el cual presentó una mala respuesta al esquema quimioterapéutico inicial. Debido al fracaso terapéutico, se solicitaron nuevas biopsias y análisis inmunohistoquímicos, siendo la muestra de cavidad oral la que confirma el diagnóstico, esta vez acertado, de linfoma linfoblástico de células T. Se modifica el esquema terapéutico a uno de mayor intensidad, logrando una evolución favorable de la enfermedad. El presente artículo corresponde al primer caso de linfoma linfoblástico de células T con localización en maxilar superior reportado en la literatura. Se recalca la dificultad diagnóstica de los variados tipos de linfomas y destaca la importancia de los exámenes complementarios para lograr un correcto diagnóstico y manejo de esta patología.

          Translated abstract

          ABSTRACT Malignant neoplasms of lymphoid origin correspond to a complex group of diseases whose diagnosis is difficult since it requires the application of immunohistochemical tests, an exhaustive analysis by the histopathological team as well as a representative sample of the tissue to be examined. We present the case of a patient previously diagnosed with non-Hodgkin's lymphoma type T, with clinical manifestation in the oral cavity, who presented a poor response to the initial chemotherapeutic scheme. Due to the therapeutic failure, new biopsies and immunohistochemical analysis were requested, being the oral cavity sample the one that confirmed the diagnosis, this time correctly, of T-cell lymphoblastic lymphoma, thus modifying the initial therapeutic scheme to one of greater intensity, achieving a favorable evolution of the disease. The present article corresponds to the first case of T-cell lymphoblastic lymphoma with maxillary location reported in the literature. It emphasizes the diagnostic difficulty of the different types of lymphomas and highlights the importance of complementary examinations to achieve a correct diagnosis and management of this pathology. We believe that this new, relatively simple technique can be an alternative to complex surgeries that involve the use of complicated bone grafts, with the consequent savings in time and morbidity for patients who associate maxillary hypoplasia and edentulism with severe bone atrophy.

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          Most cited references10

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          Lymphoblastic lymphoma: an updated review on biology, diagnosis, and treatment.

          Lymphoblastic lymphoma is a rare aggressive neoplasm of T-/B-precursors resembling acute lymphoblastic leukemia, with no or limited bone marrow involvement (<25%), that develops more frequently in children and young adults and is typically characterized by a grossly enlarged mediastinum, and whose diagnostic hallmark is the expression of a T-/B-precursor cell immunophenotype, the T-cell subset accounting for 90% of all cases. The adoption of pediatric-derived, intensive lymphoblastic leukemia-like protocols led to significantly improved results, with survival rates of about 70% and 90% in adults and children, respectively. Adequate central nervous system prophylaxis and mediastinal irradiation contributed to the therapeutic success; however, the role of radiation therapy is debated due to toxicity concerns and the excellent results obtained with radiation-free programs especially in pediatric patients. With these modern schedules, localized radiotherapy and/or hematopoietic stem cell transplants could be generally omitted, and considered only for high-risk patients identified through postinduction computed tomography/positron-emission tomography scans, minimal residual disease analysis, and new genetics and genomics. New clinical studies will have to confirm the value of these assays for risk-oriented therapy, while further therapeutic progress is expected from the introduction of new drugs and targeting agents.
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            Lymphoblastic lymphoma.

            Lymphoblastic lymphoma (LBL) is a neoplasm of immature B cells committed to the B-(B-LBL) or T-cell lineage (T-LBL) that accounts for approximately 2% of all lymphomas. Although histological features are usually sufficient to distinguish lymphoblastic from mature B- or T-cell neoplasms, of greater importance for diagnosis is the characterization of immunophenotype by flow cytometry. LBL occurs more commonly in children than in adults, mostly in males. A bone marrow involvement <25% (or 20% according to WHO) formally distinguishes LBL from ALL. The prognosis of LBL has dramatically improved with the use of intensive ALL-type chemotherapy regimens, which includes intensive intrathecal chemotherapy prophylaxis and consolidation with mediastinal irradiation. Patients with adverse prognostic features assessed by postinduction CT/positron emission tomography scans (PET) and minimal residual disease analysis (MRD) should be considered for high-dose chemotherapy and stem cell transplantation. Further therapeutic progresses are expected from the introduction of new drugs and targeting agents.
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              T-cell lymphoblastic lymphoma and leukemia: different diseases from a common premalignant progenitor?

              T-cell lymphoblastic lymphoma (T-LBL) and lymphoblastic leukemia (T-ALL) represent malignancies that arise from the transformation of immature precursor T cells. Similarities in T-LBL and T-ALL have raised the question whether these entities represent 1 disease or reflect 2 different diseases. The genetic profiles of T-ALL have been thoroughly investigated over the last 2 decades, whereas fairly little is known about genetic driver mutations in T-LBL. Nevertheless, the comparison of clinical, immunophenotypic, and molecular observations from independent T-LBL and T-ALL studies lent strength to the theory that T-LBL and T-ALL reflect different presentations of the same disease. Alternatively, T-LBL and T-ALL may simultaneously evolve from a common malignant precursor cell, each having their own specific pathogenic requirements or cellular dependencies that differ among stroma-embedded blasts in lymphoid tissues compared with solitary leukemia cells. This review aims to cluster recent findings with regard to clinical presentation, genetic predisposition, and the acquisition of additional mutations that may give rise to differences in gene expression signatures among T-LBL and T-ALL patients. Improved insight in T-LBL in relation to T-ALL may further help to apply confirmed T-ALL therapies to T-LBL patients.
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                Author and article information

                Journal
                maxi
                Revista Española de Cirugía Oral y Maxilofacial
                Rev Esp Cirug Oral y Maxilofac
                Sociedad Española de Cirugía Oral y Maxilofacial y de Cabeza y Cuello (Madrid, Madrid, Spain )
                1130-0558
                2173-9161
                June 2023
                : 45
                : 2
                : 83-87
                Affiliations
                [1] Santiago Santiago orgnameHospital San Juan de Dios orgdiv1Servicio de Cirugía Maxilofacial Chile
                [2] Santiago Santiago de Chile orgnameUniversidad de Chile orgdiv1Facultad de Odontología orgdiv2Departamento de Cirugía Maxilofacial y Traumatología Maxilofacial Chile
                Article
                S1130-05582023000200006 S1130-0558(23)04500200006
                10.20986/recom.2023.1448/2023
                78e19d19-7cf0-4a7b-a7af-4e262d8fa2c7

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.

                History
                : 10 May 2022
                : 27 June 2023
                Page count
                Figures: 0, Tables: 0, Equations: 0, References: 10, Pages: 5
                Product

                SciELO Spain

                Categories
                Casos Clínicos

                Linfoma,diagnosis,maxillofacial,T cell lymphoblastic lymphoma,Lymphoma,diagnóstico,maxilofacial,linfoma linfoblástico de células T

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