{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T04:22:54Z","timestamp":1787026974727,"version":"build-2736575974"},"reference-count":20,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,3,13]],"date-time":"2023-03-13T00:00:00Z","timestamp":1678665600000},"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. Pediatr."],"abstract":"<jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>Anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (HMGCR) myopathy is a recently recognized pathology, but appears less common in children and the characteristics of pediatric cases remain unclear.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case report<\/jats:title>\n                    <jats:p>We report a pediatric case of anti-HMGCR myopathy accompanied by skin rash. Motor function and serum creatine kinase level normalized after combinational treatment including early intravenous immunoglobulin, methotrexate, and corticosteroid.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Literature review<\/jats:title>\n                    <jats:p>We searched PubMed and identified reports with detailed clinical information of 33 pediatric patients &amp;lt;18 years old with anti-HMGCR myopathy. Among these 33 patients and our own case, skin rash and maximum serum creatine kinase level &amp;gt;5,000\u2005IU\/L were observed in 44% (15 patients) and 94% (32 patients), respectively. Skin rash was present in 15 of the 22 patients (68%) \u22657 years old and none of the 12 patients (0%) &amp;lt;7 years old. Among the 15 patients with skin rash, 12 (80%) presented with erythematous rash.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Erythematous skin rash may offer a clue to the diagnosis of anti-HMGCR myopathy in children with muscle weakness and serum creatine kinase level &amp;gt;5,000\u2005IU\/L in the absence of other myositis-specific antibodies, particularly in patients \u22657 years old. Our results suggest the importance of early anti-HMGCR testing in pediatric patients with these manifestations.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fped.2023.1102539","type":"journal-article","created":{"date-parts":[[2023,3,13]],"date-time":"2023-03-13T01:08:42Z","timestamp":1678669722000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["Case report: Diagnostic clues for anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase myopathy in pediatric patients"],"prefix":"10.3389","volume":"11","author":[{"given":"Keisuke","family":"Watanuki","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Koga","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2023,3,13]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"689","DOI":"10.1038\/s41584-020-00515-9","article-title":"Immune-mediated necrotizing myopathy: clinical features and pathogenesis","volume":"16","author":"Allenbach","year":"2020","journal-title":"Nat Rev Rheumatol"},{"key":"B2","doi-asserted-by":"publisher","first-page":"87","DOI":"10.1016\/j.nmd.2017.09.016","article-title":"224th ENMC international workshop: clinico-sero-pathological classification of immune-mediated necrotizing myopathies zandvoort, The Netherlands, 14\u201316 October 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