Literature DB >> 27358895

Myelodysplastic Syndrome with concomitant t(5;21)(q15;q22) and del(5)(q13q33): case report and review of literature.

Anup Kasi Loknath Kumar1, Brandon Weckbaugh1, Christopher Sirridge1, Janet Woodroof1, Diane Persons1, Suman Kambhampati1.   

Abstract

Chromosomal abnormalities lead to the development of hematologic malignancies such as Myelodysplastic Syndrome (MDS). Known chromosomal changes causing MDS include deletion of the long arm of chromosome 5, runt-related transcription factor 1 (RUNX1) also known as acute myeloid leukemia 1 protein (AML1), and very rarely fusion genes involving RUNX1 at t(5;21)(q15;q22). We present a case of a 71-year-old female with MDS, refractory anemia with excess blasts, type 1, with a combination of two cytogenetic abnormalities, specifically a concomitant translocation between chromosomes 5q15 and 21q22 and deletion of chromosome 5q13q33. Fluorescence in-situ hybridization (FISH) using a probe for RUNX1 (AML1), localized to 21q22, showed three FISH signals for RUNX1, consistent with rearrangement of RUNX1. Therapy was started with Lenalidomide leading to normal blood counts. Most significantly, repeat cytogenetics revealed normal karyotype and resolution of deletion on the long arm of chromosome 5 and a t(5;21). FISH negative for deletion 5q. The results altogether meet criteria for a complete cytogenetic remission (CR). We report a new case of t(5;21)(q15;q22) involving the RUNX1 gene and del(5)(q13q33) in a MDS patient, a combination of chromosomal abnormalities heretofore not reported in the literature. RUNX1 rearrangement is usually associated with an adverse prognosis in AML and MDS. Deletions of 5q are typically associated with poor prognosis in AML, however it is usually associated with a favorable prognosis in MDS. Our patient responded very well to Lenalidomide therapy with achievement of CR. Lenalidomide is approved for treatment of anemia in low and intermediate risk MDS with del (5q), however based on a search of literature it seems that RUNX1 mutations are also more prominent in patients who have responded to Lenalidomide therapy. MDS is a genomically unstable disease. Hence, it is conceivable that our patient started with a 5q minus syndrome and then acquired the second hit RUNX1 translocation leading to an accelerated phase of myeloid neoplasm or refractory anemia with excess blasts, type 1. Hence, the temporal relationship between acquisition of del 5q and RUNX1 rearrangement may have influenced the clinical outcome and possibly response to therapy.

Entities:  

Keywords:  Deletion 5q; Myelodysplastic Syndrome (MDS); lenalidomide; runt-related transcription factor 1 (RUNX1)

Year:  2016        PMID: 27358895      PMCID: PMC4923670          DOI: 10.3978/j.issn.2306-9759.2016.02.01

Source DB:  PubMed          Journal:  Stem Cell Investig        ISSN: 2306-9759


  17 in total

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Journal:  Nature       Date:  1974-10-04       Impact factor: 49.962

2.  Lenalidomide in the myelodysplastic syndrome with chromosome 5q deletion.

Authors:  Alan List; Gordon Dewald; John Bennett; Aristotle Giagounidis; Azra Raza; Eric Feldman; Bayard Powell; Peter Greenberg; Deborah Thomas; Richard Stone; Craig Reeder; Kenton Wride; John Patin; Michele Schmidt; Jerome Zeldis; Robert Knight
Journal:  N Engl J Med       Date:  2006-10-05       Impact factor: 91.245

Review 3.  The immunomodulatory agents lenalidomide and thalidomide for treatment of the myelodysplastic syndromes: a clinical practice guideline.

Authors:  Heather A Leitch; Rena Buckstein; April Shamy; John M Storring
Journal:  Crit Rev Oncol Hematol       Date:  2012-08-15       Impact factor: 6.312

4.  Single-agent lenalidomide induces complete remission of acute myeloid leukemia in patients with isolated trisomy 13.

Authors:  Todd A Fehniger; John C Byrd; Guido Marcucci; Camille N Abboud; Cheryl Kefauver; Jacqueline E Payton; Ravi Vij; William Blum
Journal:  Blood       Date:  2008-09-29       Impact factor: 22.113

5.  Trisomy 13 correlates with RUNX1 mutation and increased FLT3 expression in AML-M0 patients.

Authors:  Fernando P G Silva; Alexandra Lind; Geeske Brouwer-Mandema; Peter J M Valk; Micheline Giphart-Gassler
Journal:  Haematologica       Date:  2007-08       Impact factor: 9.941

Review 6.  The impact of translocations and gene fusions on cancer causation.

Authors:  Felix Mitelman; Bertil Johansson; Fredrik Mertens
Journal:  Nat Rev Cancer       Date:  2007-03-15       Impact factor: 60.716

7.  Chromosome pattern, occupation, and clinical features in patients with acute nonlymphocytic leukemia.

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Journal:  Cancer Genet Cytogenet       Date:  1981-11

8.  A cytogenetic study of 397 consecutive acute myeloid leukemia cases identified three with a t(7;21) associated with 5q abnormalities and exhibiting similar clinical and biological features, suggesting a new, rare acute myeloid leukemia entity.

Authors:  Eric Jeandidier; Carine Gervais; Isabelle Radford-Weiss; Estelle Zink; Catherine Gangneux; Alice Eischen; Anne Cécile Galoisy; Catherine Helias; Laurent Dano; Ornella Cammarata; Georges Jung; Inès Harzallah; Eric Guérin; Lionel Martzolff; Bernard Drénou; Bruno Lioure; Céline Tancrédi; Valérie Rimelen; Laurent Mauvieux
Journal:  Cancer Genet       Date:  2012 Jul-Aug

9.  CBFA2(AML1) translocations with novel partner chromosomes in myeloid leukemias: association with prior therapy.

Authors:  D Roulston; R Espinosa; G Nucifora; R A Larson; M M Le Beau; J D Rowley
Journal:  Blood       Date:  1998-10-15       Impact factor: 22.113

10.  Cytogenetic and molecular predictors of response in patients with myeloid malignancies without del[5q] treated with lenalidomide.

Authors:  Yuka Sugimoto; Mikkael A Sekeres; Hideki Makishima; Fabiola Traina; Valeria Visconte; Anna Jankowska; Andres Jerez; Hadrian Szpurka; Christine L O'Keefe; Kathryn Guinta; Manuel Afable; Ramon Tiu; Kathy L McGraw; Alan F List; Jaroslaw Maciejewski
Journal:  J Hematol Oncol       Date:  2012-03-05       Impact factor: 17.388

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  2 in total

Review 1.  Fragility Extraordinaire: Unsolved Mysteries of Chromosome Fragile Sites.

Authors:  Wenyi Feng; Arijita Chakraborty
Journal:  Adv Exp Med Biol       Date:  2017       Impact factor: 2.622

2.  Case Report: Personalized Therapeutical Approaches with Lenalidomide in Del(5q): A Case Series.

Authors:  Anna Stein; Anne Sophie Kubasch; Claudia Haferlach; Uwe Platzbecker
Journal:  Front Oncol       Date:  2022-03-31       Impact factor: 6.244

  2 in total

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