Acta Dermato-Venereologica 97-6 97-6CompleteContent | Page 22
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Expression of Mucin 1 in Mycosis Fungoides Tumour Cells: A Case Report
Takushi SHIRAI 1 , Yukiko KINIWA 1 *, Shiho ASAKA 2 , Yasuyo SHIMOJO 2 , Fuminao KAMIJO 1 and Ryuhei OKUYAMA 1
Department of Dermatology, and 2 Division of Clinical Investigation, Shinshu Univerisity School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano,
390-8621, Japan. E-mail: [email protected]
1
Accepted Feb 20, 2017; Epub ahead of print Feb 20, 2017
Mucin 1 (MUC1), a member of the mucin family, is a
highly glycosylated transmembrane protein normally
present on the luminal surface of secretory glands (1).
Overexpression of MUC1 in tumour cells and elevated
serum MUC1 level have been reported in patients with
cutaneous T-cell lymphoma (CTCL) (2, 3). We report
here a patient with increased serum MUC1 level and
aberrant MUC1 expression in the tumour cells of mycosis
fungoides (MF), the most common subtype of primary
cutaneous T-cell lymphoma.
CASE REPORT
A 53-year-old woman with a 5-year history of MF visited
our hospital. Her disease had progressed to tumour-stage
(T3N0M0, stage IIB) despite multiple treatments, in-
SHORT COMMUNICATION
cluding psoralen plus ultraviolet-A phototherapy, local
radiation, total-body electron-beam radiation, vorinostat
and etoposide. Serum KL-6 (Krebs von den Lungen-6,
a biomarker of interstitial pneumonia) was measured
prior to administration of intravenous interferon (IFN)-γ,
which occasionally leads to interstitial pneumonia. KL-6
is an epitope located in the N-terminal domain of MUC1.
Her serum level was high, 1,171 U/ml (normal range
105–435 U/ml) (Fig. 1A, B); however, computed tomo-
graphy (CT) showed no significant findings in her lungs,
except radiation pneumonitis in the right lung (Fig. 1C).
She was treated with IFN-γ (Day 0), followed by mo-
gamulizumab and gemcitabine. Despite these treatments,
new lesions appeared in the lymph nodes and spleen on
day 143 (T3NXM1, stage IVB). Her serum KL-6 level
increased to 9,650 U/ml as the disease progressed on
day 146. After 5 courses of CHOP (a
combination therapy of cyclophospha-
mide, doxorubicin, vincristine and
prednisolone), serum levels of soluble
interleukin-2 receptor (sIL-2R) and
KL-6 decreased, together with clinical
improvement in lymphoma lesions on
day 238. Both KL-6 and sIL-2R re-
flected the patient’s clinical condition.
Interstitial pneumonia did not occur
during the course of treatment (Fig.
1C). She underwent allogeneic bone
marrow transplantation. However, her
skin lesions worsened 14 days after the
transplantation.
Skin biopsy showed CD4-positive
lymphoma cell infiltration in both the
epidermis and dermis when the patient
was Stage IIB. MUC1 positive cells
were sparse in those lymphoma cells
by immunohistochemical staining
using a KL-6 antibody (Fig. 2A–C).
The relapsed lesions exhibited a dense
Fig. 1. Clinical characteristics and KL-6/sIL2R time course.
(A) Clinical findings of skin lesions. Day 0: initiation date of
intravenous interferon (IFN)-γ. (B) Krebs von den Lungen-6 (KL-6)
and sIL-2R levels. The patient was treated with: (i) recombinant
IFN-γ, followed by: (ii) mogamulizumab; (iii) gemcitabine; (iv)
a combination of cyclophosphamide, doxorubicin, vincristine
and prednisolone; and (v) bone marrow transplantation. (C)
Computed tomography did not reveal any significant alterations,
except for radiation pneumonitis in the right lung.
This is an open access article under the CC BY-NC license. www.medicaljournals.se/acta
Journal Compilation © 2017 Acta Dermato-Venereologica.
doi: 10.2340/00015555-2638
Acta Derm Venereol 2017; 97: 747–748