Unilateral heliotrope rash: a warning sign for anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis

Kume, Miki; Arase, Noriko; Okiyama, Naoko; Koguchi‐Yoshioka, Hanako; Tada, Tomomi; Saruban, Hiroko; Fujimoto, Manabu

doi:10.1093/rheumatology/keaa574

Cited by 8 publications

(3 citation statements)

References 7 publications

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“…Our patient also developed discoid lupus erythematosus (DLE) on the scalp, but detailed examination excluded SLE. Although a few cases of unilateral heliotrope rash have been reported in patients with dermatomyositis, 3,4 no cutaneous manifestations compatible with dermatomyositis, myositis, or interstitial lung disease, as well as myositis-specific autoantibodies, were detected in the present case. We examined CD4, CD8, CD123 positive cells in both biopsied specimens, and there was no significant difference (Table S1).…”

Section: E101contrasting

confidence: 61%

Unilateral periorbital lupus erythematosus tumidus mimicking heliotrope rash

Yamamoto

Saito

Yamamoto

2022

The Journal of Dermatology

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A 56-year-old female was referred to us, complaining of persistent erythema on the face and scalp, which had appeared, respectively, 3 months and 10 years prior. Physical examination showed edematous infiltrative erythema on the right upper eyelid (Figure 1a), as well as infiltrative scaly erythematous plaque with alopecia on the scalp (Figure 1b). Laboratory data showed normal leukocyte counts (5500/mm 3 ), as well as normal blood cell and platelet counts, and the results of blood chemistry showed normal liver and kidney function.

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Section: E101contrasting

confidence: 61%

Unilateral periorbital lupus erythematosus tumidus mimicking heliotrope rash

Yamamoto

Saito

Yamamoto

2022

The Journal of Dermatology

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“…The list of unusual manifestations of this syndrome continues to grow, including descriptions of unilateral heliotrope rash and eyelid swelling as a first sign of this phenotype [4, 5]. In a case series of 3 patients described by Kume et al [6], they also found that this unilateral heliotrope rash was only seen in DM patients with MDA-5 antibodies, preceded the usual manifestations of DM, and could be associated with rapidly progressive interstitial lung disease. Another unique early finding that has been reported in MDA-5 DM is lobular panniculitis of the cheek or mandible [7, 8].…”

Section: Discussion/conclusionmentioning

confidence: 99%

On the Nose: Anti-MDA-5 Dermatomyositis Manifesting as Perinasal Swelling

2022

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The clinical presentation of dermatomyositis (DM) is diverse, with varied phenotypes that may be correlated with specific autoantibodies. The anti-melanoma differentiation-associated gene 5 (MDA5) antibody in DM is associated with an amyopathic phenotype of DM, with several unusual cutaneous manifestation and increased risk for rapidly progressive interstitial lung disease. Initial presentation may be subtle, but early diagnosis is key to initiation of proper immunosuppressive therapy. In this report, we describe perinasal edema and erythema as a presenting complaint of anti-MDA5 DM in an otherwise healthy 40-year-old woman. The edema began shortly after heavy sun exposure and was followed by painful papules in her hands and arthritis within a few weeks. She was found to have high titer of anti-CCP and anti-MDA5, and thus was diagnosed with DM and rheumatoid arthritis overlap. A CT chest, abdomen, and pelvis showed patchy ground-glass and interstitial opacities in bilateral lower lobes consistent with mild interstitial lung disease without evidence of malignancy. Perinasal cutaneous findings and arthralgias improved with initiation of prednisone. To our knowledge, this is the first report of perinasal edema as a presenting symptom for DM and should raise suspicion for MDA-5 disease.

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“…[ 1 ] Kume et al . [ 2 ] reported three cases of unilateral heliotrope rash with ILD who tested positive for anti-MDA5 antibodies. In another report with unilateral heliotrope rash, a patient was detected positive for anti-MDA5 antibody.…”

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confidence: 99%