{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,9]],"date-time":"2026-09-09T05:08:45Z","timestamp":1788930525561,"version":"build-2803163510"},"reference-count":36,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,9,9]],"date-time":"2026-09-09T00:00:00Z","timestamp":1788912000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Antisynthetase syndrome (ASyS) is a distinct subtype of idiopathic inflammatory myopathy (IIM), characterized by multisystem involvement, including the lungs, skin, skeletal muscles, and joints, as well as the presence of anti-aminoacyl-tRNA synthetase (ARS) antibodies. While 8% to 30% of patients present with classic dermatomyositis-like rashes, atypical skin involvement can also occur. Rarely, patients may develop cutaneous edema that mimics cellulitis, which is highly susceptible to misdiagnosis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>A 72-year-old woman presented with a 1-week history of erythema, swelling, and pain in the right lower limb. Physical examination showed marked edema with a peau d\u2019orange appearance, local warmth, and tenderness. Given the detection of fungal hyphae on the right foot, cellulitis was initially suspected. However, the lesions did not improve after 5 days of cefuroxime sodium treatment, and she developed right gastrocnemius tenderness, markedly elevated muscle enzymes, and interstitial changes on chest computed tomography. Myositis antibody testing was positive for anti-melanoma differentiation-associated gene 5(anti-MDA5) and anti-histidyl-tRNA synthetase(anti-Jo-1) antibodies. Right gastrocnemius biopsy showed fiber-size variation and scattered endomysial inflammatory infiltration, without necrosis or perifascicular atrophy; immunohistochemistry showed focal\/patchy major histocompatibility complex class I (MHC-I) expression and weak non-perifascicular sarcoplasmic myxovirus resistance protein A (MxA) positivity. Antisynthetase syndrome was diagnosed. Moderate-dose oral prednisone was added and cefuroxime sodium was discontinued. Eight days later, erythema, swelling, and myalgia improved, with marked decreases in muscle enzymes and hypersensitive C-reactive protein(hs-CRP). After discharge, mycophenolate mofetil(MMF) and nintedanib were added. Three months later, the skin lesions had completely resolved, creatine kinase, lactate dehydrogenase, high-sensitivity troponin, alanine aminotransferase (ALT), and aspartate aminotransferase (AST) nearly normalized; interstitial lung disease (ILD) improved, and pleural\/pericardial effusions markedly decreased.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>Notably, this case highlights that ASyS may mimic infectious cellulitis and cause misdiagnosis. Inflammatory myopathy should be considered when atypical skin lesions are accompanied by elevated muscle enzymes and ILD.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fmed.2026.1942019","type":"journal-article","created":{"date-parts":[[2026,9,9]],"date-time":"2026-09-09T04:41:10Z","timestamp":1788928870000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Unilateral lower limb cellulitis-like edema as an atypical cutaneous manifestation of antisynthetase syndrome: a case report and targeted literature review"],"prefix":"10.3389","volume":"13","author":[{"given":"Yue","family":"Zhang","sequence":"first","affiliation":[{"name":"Department of Dermatology, Sichuan Provincial People\u2019s Hospital, School of Medicine, University of Electronic Science and Technology of China","place":["Chengdu, 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