ExpandingtheUseofRetinoidsinAcuteMyeloidLeukemia:
SpotlightonBexarotene
55Commentary on Tsai et al., p. 5619
Suzan McNamara and Wilson H. Miller, Jr.
In this issue, Tsai et al. (1) report on a phase-I clinical trial of
bexarotene in 27 non-M
3
acute myeloid leukemia (AML). AML
is characterized by abnormalities in the myeloid line at various
stages of commitment and maturation, leading to an accumu-
lation of granulocyte or monocyte precursors. The French,
American, and British classification system divided the subtypes
of AML, M
0
through M
7
, based on the stage of development of
myeloblasts at the time of diagnosis. With the exception of M
3
AML, all AML subtypes are typically treated with intensive
chemotherapy induction aimed to bring the patient into
complete hematologic remission. Eradication of residual
disease to prevent AML relapse requires consolidation therapy,
which consists of intensive chemotherapy alone or in combi-
nation with stem cell transplantation. Nevertheless, there is a
high risk of relapse, and long-term survival is less than 50%.
Furthermore, the low tolerance to intensive chemotherapy
observed in many elderly AML patients poses a treatment
challenge in a disorder that is primarily diagnosed in older
adults.
Retinoids serve as intracellular messengers or activating
ligands for the retinoic acid receptors (RAR) and retinoid X
receptors (RXR). Because of their potential to inhibit growth
and promote differentiation, retinoids hold therapeutic
promise in treating cancer, especially when used in combi-
nation with other chemotherapeutic agents. Most notably, in
acute promyelocytic leukemia patients, the M
3
AML subset,
differentiation therapy induced by all-trans -retinoic acid has
provided one of the first examples of a successful therapy
that targets the molecular cause of an aggressive malignancy
(2). Apart from acute promyelocytic leukemia, there has
been little evidence that other AML subtypes could respond
to retinoid differentiation therapy. This has been frustrating
because in vitro studies have shown more promising results.
For instance, the rexinoid bexarotene (Targretin) has been
shown to be effective in AML cell lines by inhibiting
proliferation and inducing differentiation (3). Moreover,
the combination treatment of rexinoids and cyclic AMP–
elevating drugs triggered differentiation and apoptosis in
AML patient blasts and all-trans -retinoic acid–insensitive
AML cells (4). These encouraging preclinical results promp-
ted Tsai et al. (1) to test the efficacy and safety of bexarotene
in non-M
3
AML patients.
Tsai et al. (1) report on a phase I clinical trial of bexarotene in
27 non-M
3
AML patients who had relapse, had refractory AML,
or were not eligible for chemotherapy. This study was designed
to evaluate bexarotene tolerability, toxicity, and activity.
Bexarotene was administered daily at escalating doses of 100
to 400 mg/m
2
. Overall, bexarotene was well tolerated, with
only one patient reaching dose-limiting toxicity. The most
common adverse effects were hypertriglyceridemia and hypo-
thyroidism, which were controlled with antihyperlipedemic
agents and thyroid replacement hormone, respectively. How-
ever, due to the occurrence of a grade 3 rash in three of six
patients who were treated at the highest dose of 400 mg/m
2
, the
maximum tolerated dose was determined to be 300 mg/m
2
.
Response was based on changes in peripheral blood counts and
bone marrow blast percentage. Five patients achieved a
significant clinical response, of whom four experienced a
reduction in bone marrow blasts to V5% and one patient had
a considerable reduction in blast count from 90% to 20%. Four
patients who were thrombocytopenic at the time of study
initiation had platelet responses, and seven patients had
improved neutrophil counts. Medium overall survival rate
was 3.4 months, with a range of 0.1 to 18.8+ months. After 1
year of study, three patients had continued improvements in
their blood counts.
Evidence of myeloid differentiation was identified in the
three patients with improved absolute neutrophil count and
reduction in leukemic blasts. Fluorescence in situ hybridiza-
tion analysis was done on peripheral blood granulocytes using
probes for the patients’ known leukemic cytogenetic abnor-
mality. Indeed, between 92% and 100% of the mature
circulating granulocytes held the patients’ respective cytoge-
netic abnormality. These intriguing results suggest a leukemic
origin with myeloid differentiation. Further evidence of
myeloid differentiation was observed in two patients who
developed symptoms consisting of respiratory distress, dry
cough, pleural and/or pericardial effusions, edema, and a
rapidly increasing neutrophil count. Symptoms were resolved
within 48 hours of discontinuation of bexarotene and
initiation of steroids. Interestingly, these adverse effects closely
mirror the differentiation syndrome observed in f25% of
acute promyelocytic leukemia patients treated with all-trans -
retinoic acid or arsenic trioxide. Although still not complete-
ly understood, it is thought that the syndrome is caused by
the release of cytokines from differentiating malignant
myelocytes.
Bexarotene is a synthetic rexinoid compound that has
shown efficacy in the treatment of cutaneous T-cell lympho-
CCR Translations
Authors’Affiliations: Sir Mortimer B. Davis Jewish General Hospital Segal Cancer
Center, and Department of Oncology, McGill University, Montreal, Quebec, Canada
Received 5/23/08; accepted 6/2/08.
Requestsforreprints: Wilson H. Miller, Sir Mortimer B. Davis Jewish General
Hospital Segal Cancer Center and Department of Oncology, McGill University,
3755 Cote-Ste-Catherine, E504, Montreal, Quebec, Canada PQ H3T1E2. Phone:
514-340-8222-4365; Fax: 514-340-7576; E-mail: wmiller@ldi.jgh.mcgill.ca.
F 2008 American Association for Cancer Research.
doi:10.1158/1078-0432.CCR-08-1081
www.aacrjournals.org ClinCancerRes2008;14(17)September1,2008 5311
Research.
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