Volume 1 • Issue 3 • 1000118
J Leuk
ISSN: 2329-6917 JLU, an open access journal
Open Access Case Report
Achkar et al., J Leuk 2013, 1:3
DOI: 10.4172/2329-6917.1000118
*Corresponding author: Walid Al-Achkar, Human Genetics Division, Department
of Molecular Biology and Biotechnology , Atomic Energy Commission of Syria, 17th
Nissan St., Kafersouseh, Damascus, 6091, Syria, Tel: 963-11-2132580; Fax: 963-
11-6112289; E-mail: ascientifc@aec.org.sy , i8lith@mti.uni-jena.de
Received May 22, 2013; Accepted July 18, 2013; Published July 22, 2013
Citation: Achkar WAL, Moassass F, Ikhtiar A, Othman MAK, Liehr T, et al. (2013)
Cytogenetic Evolution in a Patient with Chronic developing a Secondary Acute
Myelogenous Leukemia Subtype M5 Resistant to Imatinib Mesylate Therapy. J
Leuk 1: 118. doi:10.4172/2329-6917.1000118
Copyright: © 2013 Achkar WAL, et al. This is an open-access article distributed
under the terms of the Creative Commons Attribution License, which permits
unrestricted use, distribution, and reproduction in any medium, provided the
original author and source are credited.
Abstract
Here we report an unusual case of Chronic Myelogeneous Leukemia (CML) developing towards an Acute
Myelogeneous Leukemia subtype M5 (AML-M5). The chromosomal constitution was at (fnal) stage of AML-M5:
Philadelphia chromosome positivity with multiple trisomies, a double t (9; 22) (q34; q11) and an AML1/MDS1/EVI1
(AME) fusion transcript resulting from a t (3; 21) (q26; q22). The latter translocation was detectable frst in blast
phase of CML and remained present in AML-M5 stage. Overall, four chromosomal analyses were done within 19
months, describing the ongoing karyotypic evolution during this transformation. Unfortunately this exceptional patient
did not respond to Imatinib- (IM) or Nilotinib-therapy. These fnding May by a frst hint that CML-patients acquiring a
t (3; 21) (q26; q22) might be appropriate to bone marrow transplantation rather than for IM-therapy.
including a double t (9; 22) (q34; q11) and an AME fusion transcript
resulting from a t(3;21)(q26;q22).
Case Report
A 27-year old male was diagnosed with CML in chronic phase in
March 2010 due to splenomegaly. In June 2011, the patient presented
for the third time (Table 1) with a WBC of 4.3×10
9
/l consisting of 48.6%
neutrophils, 27.7% lymphocytes and 23.7% monocytes. Te platelets
count was 55×10
9
/l and the hemoglobin level was 10.1 g/dl. Te serum
LDH level was 1024 U/l and the serum Alkaline Phosphatase (ALP)
level was 301 U/l (normal level up to 300 U/l). Te patient was treated
with Imatinib mesylate (IM) at 400 mg/day for overall 15 months in the
total; treatment regime was not altered as still the clinicians expected
to have a positive efect from it even afer blastic change was evident.
In October 2011, the patient presented for the fourth time, having a
WBC of 4.4×10
9
/l consisting of 63% neutrophils, 28.4% lymphocytes
and 8.4% monocytes (Table 1). Te platelets count was 70×10
9
/l and
the hemoglobin level was 12 g/dl. Te serum LDH level was 900 U/l.
Te patient was treated with Nilotinib at 800 mg/day for overall 4
months. In November 2011, he passed away under the treatment due
to unknown reasons.
Karyotyping was performed one time before, two times during IM
treatment and one time under Nilotinib treatment (Table 1). Initially a
CML-specifc karyotype 46, XY, t (9; 22) (20) was found, which evolved
Keywords: Chronic myeloid leukemia (CML); Acute myeloid
leukemia (AML); Trisomy 9; Fluorescence in situ hybridization (FISH);
Imatinib resistant
Introduction
Te Philadelphia (Ph) chromosome is a common cytogenetic
abnormality in hematologic malignancies. Although it is most
frequently associated with Chronic Myelogeneous Leukemia (CML),
it can also be present in Adult Precursor B-Lymphoblastic Leukemia/
Lymphoma (ALL) and less commonly in pediatric and Adult Acute
Myelogeneous Leukemia (AML) [1]. At the molecular level, the t (9;
22) (q34; q11) juxtaposes the 5’ end of the breakpoint cluster region
(BCR) gene on chromosome 22 to the 3’ end of the Abelson Tyrosine
Kinase (ABL) gene on chromosome 9. CML in blast crisis is ofen
accompanied by the presence of additional chromosomal aberrations
[1]. Amongst those, activation of the EVI1 (Ectopic Viral Integration
Site 1) gene has been reported in a small percentage of patients;
ectopic expression of the EVI1 gene is usually due to recurrent 3q26
rearrangements such as the t(3;21)(q26;q22) and the inv(3;3)(q21q26)
[1]. Te t (3;21) (q26;q22) resulting in the AML1/MDS1/EVI1 (AME)
fusion is reported in t-AML, t-MDS (i.e. myelodysplastic syndrome)
and in CML [2]. In this translocation parts of the AML1 gene could
be fused to one of the two aforementioned genes which are adjacent
in 3q26. Tese fusion products, in cooperation with other genetic
abnormalities, are capable of blocking myeloid diferentiation possibly
by interfering with the normal transcriptional regulatory functions of
AML1. Furthermore, they initiate malignant transformation in cell
lines and myeloid leukemia in mouse tumor models. However, the
clinical features of t(3;21)-associated human leukemia have not been
well defned to date due to the lack of any large clinical case studies [3].
Te association of acquired trisomies involving various
chromosomes and hematological malignancies is well established.
Te presence of some single acquired autosomal trisomies may be
indicative for prognosis, too [4]. Trisomies 9 and 10 are rare observed
in AML [4,5].
Here we reported a Ph chromosome positive Acute Myelogeneous
Leukemia Subtype M5 (AML-M5) which transformed from a Chronic
Myelogeneous Leukemia (CML) presenting multiple trisomies,
Cytogenetic Evolution in a Patient with Chronic developing a Secondary
Acute Myelogenous Leukemia Subtype M5 Resistant to Imatinib Mesylate
Therapy
Walid AL Achkar
1
*, Faten Moassass
1
, Adnan Ikhtiar
2
, Moneeb AK Othman
3
, Thomas Liehr
3
and Abdulsamad Wafa
1
1
Human Genetics Division, Flow-cytometry Laboratory; Department of Molecular Biology and Biotechnology, Atomic Energy Commission of Syria, Damascus, Syria
2
Mammalians Biology Division, Flow-cytometry Laboratory; Department of Molecular Biology and Biotechnology, Atomic Energy Commission of Syria, Damascus, Syria
3
Jena University Hospital, Institute of Human Genetics, Jena, Germany
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ISSN: 2329-6917