Acta Dermato-Venereologica 99-9CompleteContent | Page 17
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SHORT COMMUNICATION
Diagnosis of Mycosis Fungoides Following Administration of Dupilumab for Misdiagnosed Atopic
Dermatitis
Takahito CHIBA, Takuya NAGAI, Shin-ichi OSADA and Motomu MANABE
Department of Dermatology and Plastic Surgery, Akita University Graduate School of Medicine, 1-1-1 Hondo, Akita 010-8543, Japan. E-mail:
[email protected]
Accepted Apr 30, 2019; E-published May 2, 2019
The effect of anti-interleukin (IL)-4/-13 treatment, du
pilumab, on allergic diseases and the resulting various
unexpected phenomena have been reported in the lite
rature. The clinical features of mycosis fungoides (MF)
with multiple erythematous lesions are similar to those of
atopic dermatitis (AD). Thus, anti-IL-4/-13 therapy may
be erroneously administered to patients with MF with
features similar to those of AD. This report is the first to
describe a case of MF diagnosed after administration of
dupilumab for AD.
CASE REPORT
A 58-year-old man had had AD since early childhood. Although
remission had occurred in adolescence, eczema had appeared on
his trunk and limbs at the age of 47 years. He presented to a der
matologist and was administered topical corticosteroid ointment,
betamethasone butyrate propionate, based on the diagnosis of
recurrent AD. In 2013 the patient was referred to our university
for treatment of a carcinoma of the maxillary sinus using chemo
radiotherapy. Treatment was successful. Subsequently, the patient
continued treatment for AD in our department. The cutaneous erup
tion was somewhat controlled by topical treatments; however, at
times he experienced intense itching (Fig. 1). He reported a history
of allergic rhinitis, conjunctivitis and asthma. Given the severity of
his AD, treatment with dupilumab 600 mg, subcutaneously was ini
tiated on the first day, followed by 300 mg every 2 weeks in 2018.
Serum examination prior to administration revealed elevated levels
of thymus-and activation-regulated chemokine (TARC, 4,634 pg/
ml; normal range: < 450 pg/ml), IgE (1,280 IU/ml; normal range:
< 250 IU/ml), and lactate dehydrogenase (LDH, 269 U/l; normal
range: 124–224 U/l). The eosinophil count was within normal
Fig. 1. (a) Erythematous lesions on the face. (b) Poikiloderma lesions
accompanied by papules and nodules on the back. Written permission
from the patient is given to publish these photos.
doi: 10.2340/00015555-3208
Acta Derm Venereol 2019; 99: 818–819
Fig. 2. (a) Face and (b) back after 1 month of treatment with dupilumab.
Written permission from the patient is given to publish these photos.
limits. The Eczema Area and Severity Index (EASI) was 34.4,
while the Dermatology Life Quality Index (DLQI) was 8. During
treatment with dupilumab, the faint erythematous lesions on the
face and trunk became more obvious, despite continuous applica
tion of the topical steroid (Fig. 2). After 1 month, the EASI and
DLQI were slightly decreased (29 and 6, respectively); however,
there was no improvement in pruritic symptoms. Laboratory tests
revealed that TARC, IgE, and LDH levels were 3,948 pg/ml, 924
IU/ml, and 287 U/l, respectively. Owing to exacerbation of the ery
thematous lesions, skin biopsy of the erythematous macule on the
trunk was performed. Histopathology showed band-like atypical
lymphocytic infiltration into the dermis. Atypical epidermotropic
lymphocytes without spongiosis were observed in the epidermis,
which formed Pautrier’s microabscess (Fig. 3). Almost all of these
Fig. 3. Histopathology of the biopsy specimen from the erythematous
lesion on the chest shows lymphocytic infiltrate in the dermis with
prominent epidermotropism (a: haematoxylin and eosin; H&E ×100)
and atypical lymphocytes into the epidermis (b: H&E ×400).
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Journal Compilation © 2019 Acta Dermato-Venereologica.