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INVESTIGATIVE REPORT
The MicroRNA Expression Profile Differs Between Erythrodermic
Mycosis Fungoides and Sézary Syndrome
Anne Hald RITTIG 1 , Lise Maria LINDAHL 1 , Claus JOHANSEN 1 , Pamela CELIS 2 , Niels ØDUM 3 , Lars IVERSEN 1 and Thomas
LITMAN 3
Department of Dermatology, Aarhus University Hospital, 2 Department of Molecular Medicine, Aarhus University, Aarhus, and 3 Leo Foundation
Skin Immunology Research Center; Department of Immunology and Microbiology, University of Copenhagen, Copenhagen, Denmark
1
It is difficult to distinguish erythrodermic mycosis
fungoides from Sézary syndrome due to their similar
clinical and histological features. The main purpose of
this study was to investigate whether microRNA ex-
pression profiles in lesional skin could discriminate pa-
tients with erythrodermic mycosis fungoides from those
with Sézary syndrome. A further aim was to assess
whether the microRNA expression profiles in erythro-
dermic mycosis fungoides skin was more comparable
to microRNA expression profiles of Sézary syndrome
or early-stage mycosis fungoides. RNA was extracted
from diagnostic skin biopsies, followed by quantita-
tive reverse transcription polymerase chain reaction
analysis of 383 microRNAs. Twenty-seven microRNAs
were significantly differentially expressed between
erythrodermic mycosis fungoides and Sézary syndro-
me. Moreover, erythrodermic mycosis fungoides sho-
wed microRNA features overlapping with Sézary syn-
drome and early-stage mycosis fungoides, although
hierarchical cluster analysis co-clustered erythro-
dermic mycosis fungoides with early-stage mycosis
fungoides rather than with Sézary syndrome. These
findings underscore that erythrodermic mycosis
fungoides and Sézary syndrome are different diseases.
Key words: mycosis fungoides, Sézary syndrome, cutaneous T-
cell lymphoma, microRNA.
Accepted Aug 26, 2019; E-published Aug 27, 2019
Acta Derm Venereol 2019; 99: 1148–1153.
Corr: Anne Hald Rittig, Department of Dermatology, Aarhus University
Hospital, Palle Juul-Jensens Boulevard 99, E201, DK-8200 Aarhus N, Den-
mark. E-mail: [email protected]
C
utaneous T-cell lymphoma (CTCL) is a group of rare
heterogeneous lymphoproliferative disorders prima-
rily confined to the skin. The most prevalent clinical form
of CTCL is mycosis fungoides (MF) (1, 2). Early-stage
MF comprises patch and plaque skin lesions, whereas
advanced stages involve skin tumours and erythrodermic
MF (eMF) (3, 4). eMF and the more aggressive leukae-
mic variant, Sézary syndrome (SS), are characterized by
chronic erythroderma with ≥ 80% skin involvement (4).
eMF and SS are difficult to distinguish because of their
similar clinical features, with erythroderma and lymph
adenopathy combined with symptoms such as pruritus,
skin burning, and chills (5). However, eMF sometimes
progresses through patch- and/or plaque-stage disease
doi: 10.2340/00015555-3306
Acta Derm Venereol 2019; 99: 1148–1153
SIGNIFICANCE
Erythrodermic mycosis fungoides and Sézary syndrome
share clinical and histological features, making it difficult to
distinguish these diseases. It has been discussed previous-
ly whether erythrodermic mycosis fungoides and Sézary
syndrome are different stages of the same disease. How
ever, differences in treatment and prognoses indicate that
the diseases should be considered separately. MicroRNAs
are small sequences of RNA, which have the potential to
discriminate clinically similar diseases. This study showed
that 27 microRNAs discriminated erythrodermic mycosis
fungoides from Sézary syndrome. These data support the
perception of erythrodermic mycosis fungoides and Sézary
syndrome as different diseases.
and has various levels of blood involvement, whereas SS
usually presents with erythroderma and significant blood
involvement (6, 7). Controversies still exist regarding
whether MF and SS are distinct diseases or different
manifestations of a single disease (8, 9), which has led
to substantial discussions about the distinction between
eMF and SS (6, 9, 10). Discrimination between SS and
eMF is essential due to differences in treatment recom-
mendations and prognosis (7, 11, 12).
MicroRNAs (miRNAs) may have the potential to
discriminate between SS and eMF. miRNAs are short
non-coding RNA molecules that regulate gene expres-
sion by modulating translation of messenger RNA (13).
Thus, miRNAs are involved in important biological
functions, and altered regulation of miRNAs plays a key
role in cancer development, progression and metastasis
(14). Many miRNAs are differentially expressed in
CTCL compared with normal skin (15–17). They have
been proposed to discriminate CTCL from benign skin
diseases as a diagnostic marker (18–20), and a prognostic
3-miRNA classifier was developed recently for patients
diagnosed with early-stage MF (21). Moreover, single
miRNAs (e.g. miR-155) may have essential regulatory
functions in CTCL (22, 23), and a drug targeting miR-
155, cobomarsen, was developed recently and tested in
a clinical trial with promising outcomes (24).
This study examined the miRNA expression profiles
of diagnostic lesional skin biopsies from early-stage MF,
eMF, and SS, with the aim of: (i) examining whether
a distinct miRNA expression profile can discriminate
eMF from SS; and (ii) addressing whether the miRNA
This is an open access article under the CC BY-NC license. www.medicaljournals.se/acta
Journal Compilation © 2019 Acta Dermato-Venereologica.