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Focal hyperkeratosis overlying
the palmar faces of
revealing a juvenile
Dermatomyositis (DM) is an idiopathic inﬂammatory myopathy
characterized by an inﬂammatory inﬁltrate primarily affecting
the skeletal muscle and skin.
It is the commonest of the idio-
pathic inﬂammatory myopathies of childhood, comprising 85%
of cases. It has an annual incidence estimated to range between
1.9 and 4.1 per million children.
Euwer proposed a classiﬁca-
tion in which DM skin manifestations are subdivided into
pathognomonic, highly characteristic and compatible skin
The most common and peculiar manifestations include
Gottron’s papules, Gottron’s sign and heliotrope rash. Recogni-
tion of cutaneous and histopathologic ﬁndings in DM is essen-
tial for prompt and accurate diagnosis and treatment of DM.
A 2-year-old girl presented with a four-month history of
recurrent palmar lesions. A hand-foot-and-mouth disease was
eliminated because of persistence of the lesions. The patient was
treated in a second time with topical corticosteroids for pre-
sumptive diagnosis of acropustulosis. In the same time, the
patient presented with a lameness and was plastered in a context
of fall. Dermatological examination revealed focal hyperkeratosis
overlying the palmar faces of interphalangeal and metacarpopha-
langeal joints (Fig. 1). Similar lesions were observed on the helix
and the big toe. After removing the plaster, the patient still
limped and complained of a painful ankle. No fever nor warmth
or oedema of the child’s ankles or other joints was noted. One
month later, heliotropic rash and Gottron’s sign appeared. This
was suggestive of atypical juvenile DM. Inﬂammatory pain of
the left ankle was retained without arthritis. Muscular testing
did not ﬁnd muscle weakness. The laboratory ﬁndings revealed
elevated serum levels of lactate dehydrogenase at 884 IU/L (nor-
mal range 0–145 IU/L) and slightly elevated levels of aspartate
transaminase at 66 U/L. Creatine phosphokinase (CK) was nor-
mal. No antinuclear antibodies or anti-extractable nuclear anti-
gens including anti-Jo-1 antibody were found. Histological
examination of a skin biopsy specimen from the right hand
showed a slight epidermal hyperplasia, some scattered necrotic
keratinocytes in the midpart of the epidermis, focal interface
dermatitis and dilated vessels within the superﬁcial dermis
Fig. 2). Muscle biopsy was not performed. The whole-body
MRI did not demonstrate signs of myositis. Inﬂammation in
proximal muscles was assessed by (18)F FDG-PET-CT. Two
months later, levels of CK were elevated at 307. She was treated
with oral prednisolone 0.5 mg/kg/day and methotrexate 7.5 mg/
week. Currently, skin lesions, including focal hyperkeratosis, dis-
appeared, whereas Gottron’s sign was ﬂuctuant. Weakness of the
muscle strength improved with a normal CK level.
The case presentation is unique when compared with the
commonly reported ﬁndings of JDM. The presence of long-
lasting focal hyperkeratosis overlying the palmar face of
interphalangeal and metacarpophalangeal joints is clearly dif-
ferent from typical lesions of the ﬁngers observed in JDM.
These palmar lesions differ from Wong dermatomyositis.
This is a rarely described phenomenon in which patients
with DM develop cutaneous hyperkeratotic lesions including
diffuse palmoplantar hyperkeratosis and follicular hyperker-
atosis that resemble pityriasis rubra pilaris and histologically
show follicular hyperkeratosis and hair follicle destruction.
Our observation is also different from mechanic’s hands
during antisynthetase syndrome.
Our observation also suggests that [(18)F]FDG-PET/CT is
useful in the diagnosis of polymyositis/DM when whole MRI
failed in demonstrating myositis.
The utility of LDH in normal
creatine kinase dermatomyositis has also to be noted.
Figure 1 Focal hyperkeratosis overlying the palmar faces of inter-
phalangeal and metacarpophalangeal joints.
© 2017 European Academy of Dermatology and Venereology
2018, 32, e86–e121
Letters to the Editor