Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutation

Familial platelet disorder with propensity to develop acute myeloid leukemia (FPD/AML) is a rare autosomal dominant disorder caused by inherited mutation of RUNX1. To date only 35 families have been described. We report on a family in which number and function of platelet were impaired in members of...

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Main Authors: V. O. Bobrynina, O. Yu. Baranova, E. V. Samochatova1, A. A. Maschan
Format: Article
Language:Russian
Published: ABV-press 2014-07-01
Series:Онкогематология
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Online Access:https://oncohematology.abvpress.ru/ongm/article/view/77
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author V. O. Bobrynina
O. Yu. Baranova
E. V. Samochatova1,
A. A. Maschan
author_facet V. O. Bobrynina
O. Yu. Baranova
E. V. Samochatova1,
A. A. Maschan
author_sort V. O. Bobrynina
collection DOAJ
description Familial platelet disorder with propensity to develop acute myeloid leukemia (FPD/AML) is a rare autosomal dominant disorder caused by inherited mutation of RUNX1. To date only 35 families have been described. We report on a family in which number and function of platelet were impaired in members of 4 generations. Acute myeloid leukemia developed in 3 members of 2 generations. The age of leukemia development was 11, 19 и 76 years. In all 5 affected family members available for study novel heterozygous mutation in runt domain of RUNX1 (c2018 A > C, pT147P) was detected by direct sequencing. This mutation is predicted to impair binding of CBF to core motif of DNA. Two affected patients died of leukemia; while in one complete remission was achieved with conventional and consolidated with allogeneic BMT from HLA-matched sister proved to be RUNX1 mutation negative.
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institution Kabale University
issn 1818-8346
2413-4023
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publishDate 2014-07-01
publisher ABV-press
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series Онкогематология
spelling doaj-art-b709feac727a40d69b4f2047e03600cf2025-08-20T03:35:33ZrusABV-pressОнкогематология1818-83462413-40232014-07-016461110.17650/1818-8346-2011-6-4-6-1192Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutationV. O. Bobrynina0O. Yu. Baranova1E. V. Samochatova1,2A. A. Maschan3Federal Research Center of Pediatric Hematology, Oncology and ImmunologyN.N. Blokhin Russian Cancer Research Center, Russian Academy of Medical SciencesFederal Research Center of Pediatric Hematology, Oncology and ImmunologyFederal Research Center of Pediatric Hematology, Oncology and ImmunologyFamilial platelet disorder with propensity to develop acute myeloid leukemia (FPD/AML) is a rare autosomal dominant disorder caused by inherited mutation of RUNX1. To date only 35 families have been described. We report on a family in which number and function of platelet were impaired in members of 4 generations. Acute myeloid leukemia developed in 3 members of 2 generations. The age of leukemia development was 11, 19 и 76 years. In all 5 affected family members available for study novel heterozygous mutation in runt domain of RUNX1 (c2018 A > C, pT147P) was detected by direct sequencing. This mutation is predicted to impair binding of CBF to core motif of DNA. Two affected patients died of leukemia; while in one complete remission was achieved with conventional and consolidated with allogeneic BMT from HLA-matched sister proved to be RUNX1 mutation negative.https://oncohematology.abvpress.ru/ongm/article/view/77family thrombocytopenia/thrombocytopathiaacute myeloid leukemiarunx1
spellingShingle V. O. Bobrynina
O. Yu. Baranova
E. V. Samochatova1,
A. A. Maschan
Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutation
Онкогематология
family thrombocytopenia/thrombocytopathia
acute myeloid leukemia
runx1
title Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutation
title_full Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutation
title_fullStr Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutation
title_full_unstemmed Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutation
title_short Family platelet disorder with propensity to acute myeloid leukemia: new family with RUNX1 mutation
title_sort family platelet disorder with propensity to acute myeloid leukemia new family with runx1 mutation
topic family thrombocytopenia/thrombocytopathia
acute myeloid leukemia
runx1
url https://oncohematology.abvpress.ru/ongm/article/view/77
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AT evsamochatova1 familyplateletdisorderwithpropensitytoacutemyeloidleukemianewfamilywithrunx1mutation
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