ARTICLE
Auteur(s) : Yasuhiro Kawachi, Enmi
Hori, Michiko Itoh, Yasuhiro Fujisawa, Junichi Furuta, Yasuhiro
Nakamura, Yoshiyuki Ishii, Fujio Otsuka
Department of Dermatology, Institute of Clinical
Medicine, University of Tsukuba, 1-1-1, Tennodai, Tsukuba,
Ibaraki 305-8575, Japan
A 32-year-old woman developed reddish macules on both eyelids
and pruriginous edematous erythematous lesions on the shoulders and
thighs. The patient also showed erythematous cutaneous lesions on
the bilateral elbows and knees, bilateral extensor sites of the
upper aspects of the arms, and the periungual nailfolds. Four
months later, she presented with proximal muscular pain and
weakness that became gradually worse. On referral to our clinic,
physical examination revealed fluctuating cystic soft tissue
swelling with superficial scaling erythema on the bilateral elbows
(figure 1C) in
addition to the typical observations of heliotrope periorbital
edema (figure
1A), Gottron’s papules (figure 1B). The
subcutaneous cystic masses showed slight tenderness but no
spontaneous pain. The patient had not been aware of any injury or
unusual mechanical stress on the affected elbows. Laboratory data
indicated a slight increase in serum aldolase level
(7.0 IU/dL; normal range, 1.7-5.7 IU/dL) with a normal
serum level of creatine phosphokinase (CK) and moderately elevated
C-reactive protein level (2.2 mg/dL). The patient was negative
for anti-nuclear and anti-Jo-1 antibodies. A skin biopsy
obtained from the erythematous eruption on the right elbow revealed
slight liquefaction degeneration in the basal layer of the
epidermis, and massive mononuclear cell infiltration was noted in
fat lobules in subcutaneous layers. No apparent necrotizing
vasculitis or crystal deposition was seen throughout the whole
specimen. Magnetic resonance imaging (MRI) of the elbows revealed a
bursa markedly distended by fluid and rim enhancement, which
indicated inflammation around the bursa wall (figures 1D, E). Aspirated
fluid from the lesion was negative on Gram staining. Repeated
bursal fluid cultures of aspirated fluids yielded no microbial
pathogens and revealed the bursitis of both elbows to be aseptic.
Based on these findings, a diagnosis of dermatomyositis with
aseptic olecranon bursitis was made and oral prednisolone was
started at a dose of 50 mg daily. The patient showed rapid
improvement of the eruptions, including the heliotrope periorbital
edema and the Gottron’s papules, and steroid therapy resulted in a
gradual decrease of redness and swelling due to the bursitis, and
the serum aldolase level decreased to within the normal range,
which allowed gradual tapering of the steroid dose. Four months
after the initial treatment, the symptoms of bursitis at both
elbows disappeared completely and MRI revealed no cystic masses or
fluid retention in either elbow.
The most common cause of aseptic olecranon bursitis is chronic
repetitive trauma with direct pressure on the olecranon, but with
the exception of rheumatoid arthritis cases, aseptic bursitis
associated with autoimmune connective tissue diseases is quite
rare, and there have been no previous reports of aseptic bursitis
associated with dermatomyositis. The skin manifestations are the
distinguishing clinical features of dermatomyositis, with
well-known primary classic cutaneous lesions of heliotrope
periorbital rash, Gottron’s sign, atrophic poikilodermatous
erythema, pruritic flagellate erythema, periungual telangiectasia,
and photosensitivity. However, subcutaneous involvement during the
course of this disease is uncommon. Previous reports described
subcutaneous involvement, including panniculitis [1], vasculitis
[2], massive calcinosis [3], and generalized edema [4, 5], as rare
skin manifestations of dermatomyositis and indicators of poor
prognosis. Here, we described a patient with dermatomyositis
showing aseptic olecranon bursitis as a subcutaneous
manifestation.
The present case showed panniculitis without vasculitis as well
as bursitis in the surrounding subcutaneous tissue of the olecranon
bursa. As MRI examination revealed bursal membrane inflammation
without surrounding panniculitis (figures 1D, E;
arrowheads), we feel that the bursitis arose first in this case and
the panniculitis of the adjacent fat tissue occurred as a secondary
effect due to extension of the inflammation. In summary, the
observations in the present case indicated that aseptic effusive
bursitis is a rare but cautionary manifestation of
dermatomyositis.
Acknowledgements
Financial support: none. Conflict of interest: none
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