{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,7]],"date-time":"2026-06-07T17:27:42Z","timestamp":1780853262266,"version":"3.54.1"},"reference-count":43,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2024,7,31]],"date-time":"2024-07-31T00:00:00Z","timestamp":1722384000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,1,16]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>L\u00f6fgren syndrome (LS) is a sarcoidosis subtype characterised by an acute disease course, bilateral hilar lymphadenopathy (BHL), erythema nodosum (EN), and ankle arthritis. LS in Caucasians appears to be self-limiting; however, our patients require glucocorticoid (GC) treatment. Here, we present a case of LS and review the literature to identify the characteristics of Japanese patients with LS. A 66-year-old woman was referred to Kobe City Medical Center General Hospital; she initially presented with an acute onset of low-grade fever and ankle arthritis, followed by EN. Skin biopsy revealed a noncaseating granuloma, and a chest computed tomography scan displayed BHL; she was diagnosed with LS. Her arthralgia ameliorated spontaneously, but the erythema persisted, necessitating GC treatment. A literature review revealed that the Japanese LS patients showed more fever, were more frequently treated with GCs, and more patients seemed to relapse, which may be explained by the absence of human leucocyte antigen\u2013DR isotype 3, a good prognostic allele in Caucasians. Japanese LS may cause severe symptoms after development because of the differences in human leucocyte antigens from foreign countries. For early diagnosis, it is important to evaluate EN and BHL in patients with polyarthritis involving ankle arthralgia.<\/jats:p>","DOI":"10.1093\/mrcr\/rxae034","type":"journal-article","created":{"date-parts":[[2024,8,1]],"date-time":"2024-08-01T01:48:09Z","timestamp":1722476889000},"page":"193-199","source":"Crossref","is-referenced-by-count":3,"title":["L\u00f6fgren syndrome, characteristics of Japanese cases: A case and a review of the literature"],"prefix":"10.1093","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-7894-6389","authenticated-orcid":false,"given":"Hirokazu","family":"Taguchi","sequence":"first","affiliation":[{"name":"Department of Rheumatology, Kobe City Medical Center General Hospital , Hyogo,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shuji","family":"Sumitomo","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Kobe City Medical Center General Hospital , Hyogo,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9810-7644","authenticated-orcid":false,"given":"Hideki","family":"Oka","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Kobe City Medical Center General Hospital , Hyogo,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shigeo","family":"Hara","sequence":"additional","affiliation":[{"name":"Department of Diagnostic Pathology, Kobe City Medical Center General Hospital , Hyogo,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2927-9159","authenticated-orcid":false,"given":"Koichiro","family":"Ohmura","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Kobe City Medical Center General Hospital , Hyogo,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2024,7,31]]},"reference":[{"key":"2025011615465481800_R1","doi-asserted-by":"crossref","first-page":"424","DOI":"10.1111\/j.0954-6820.1953.tb07039.x","article-title":"Primary pulmonary sarcoidosis. I. Early signs and symptoms","volume":"145","author":"Lofgren","year":"1953","journal-title":"Acta Med Scand"},{"key":"2025011615465481800_R2","doi-asserted-by":"crossref","first-page":"465","DOI":"10.1111\/j.0954-6820.1953.tb07044.x","article-title":"Primary pulmonary sarcoidosis. II. Clinical course and prognosis","volume":"145","author":"Lofgren","year":"1953","journal-title":"Acta Med Scand"},{"key":"2025011615465481800_R3","first-page":"736","article-title":"Statement on sarcoidosis. Joint Statement of the American Thoracic Society (ATS), the European Respiratory Society (ERS) and the World Association of Sarcoidosis and Other Granulomatous Disorders (WASOG) adopted by the ATS Board of Directors and by the ERS Executive Committee, February 1999","volume":"160","year":"1999","journal-title":"Am J Respir Crit Care Med"},{"key":"2025011615465481800_R4","doi-asserted-by":"crossref","first-page":"240","DOI":"10.1016\/S0002-9343(99)00223-5","article-title":"Lofgren\u2019s syndrome revisited: a study of 186 patients","volume":"107","author":"Mana","year":"1999","journal-title":"Am J Med"},{"key":"2025011615465481800_R5","doi-asserted-by":"crossref","first-page":"499","DOI":"10.1136\/ard.61.6.499","article-title":"Sarcoid arthritis: clinical characteristics, diagnostic aspects, and risk factors","volume":"61","author":"Visser","year":"2002","journal-title":"Ann Rheum Dis"},{"key":"2025011615465481800_R6","doi-asserted-by":"crossref","first-page":"101","DOI":"10.1002\/art.1780130201","article-title":"Periarticular inflammation, bilateral hilar adenopathy and a sarcoid reaction","volume":"13","author":"Caplan","year":"1970","journal-title":"Arthritis Rheum"},{"key":"2025011615465481800_R7","doi-asserted-by":"crossref","first-page":"141","DOI":"10.1016\/j.jbspin.2019.10.001","article-title":"Sarcoidosis presenting with and without Lofgren\u2019s syndrome: clinical, radiological and behavioral differences observed in a group of 691patients","volume":"87","author":"Rubio-Rivas","year":"2020","journal-title":"Joint Bone Spine"},{"key":"2025011615465481800_R8","doi-asserted-by":"crossref","first-page":"372","DOI":"10.1183\/09031936.00075307","article-title":"Epidemiology of sarcoidosis in Japan","volume":"31","author":"Morimoto","year":"2008","journal-title":"Eur Respir 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syndrome","volume":"40","author":"Higashimoto","year":"2002","journal-title":"AJRS"},{"key":"2025011615465481800_R13","first-page":"207","article-title":"A case of sarcoidosis with rheumatic features (L\u00f6fgren\u2019s syndrome)","volume":"41","author":"Niimi","year":"2003","journal-title":"Nihon Kokyuki Gakkai Zasshi = J Jpn Respir Soc"},{"key":"2025011615465481800_R14","doi-asserted-by":"crossref","first-page":"534","DOI":"10.2169\/internalmedicine.42.534","article-title":"Sarcoidosis with multiple organ involvement emerging as Lofgren\u2019s syndrome","volume":"42","author":"Oshima","year":"2003","journal-title":"Intern Med"},{"key":"2025011615465481800_R15","first-page":"761","article-title":"A male case of acute sarcoidosis with fever, polyarthralgia, erythema nodosum, and bilateral hilar lymphadenopathy: L\u00f6fgren\u2019s syndrome","volume":"43","author":"Izumo","year":"2005","journal-title":"Nihon Kokyuki Gakkai Zasshi = J Jpn Respir 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L\u00f6fgren\u2019s syndrome with acute aggravation of pulmonary lesion","volume":"61","author":"Kosaka","year":"2007","journal-title":"J Clin Dermatol"},{"key":"2025011615465481800_R20","first-page":"43","article-title":"[A case of L\u00f6fgren\u2019s syndrome, presented with bilateral ankle arthritis] Ryosokuashikansetuen wo syusoni touka wo zyusin sita L\u00f6fgren syokogun no itirei","volume":"42","author":"Hama","year":"2009","journal-title":"Proc Kanto Rheumatol Conf"},{"key":"2025011615465481800_R21","first-page":"1017","article-title":"A case of L\u00f6fgren\u2019s syndrome","volume":"64","author":"Risa","year":"2010","journal-title":"Jpn J Clin Dermatol"},{"key":"2025011615465481800_R22","doi-asserted-by":"crossref","first-page":"113","DOI":"10.7878\/jjsogd.32.113","article-title":"A case of nodular muscular sarcoidosis with L\u00f6fgren\u2019s syndrome","volume":"32","author":"Takasa","year":"2012","journal-title":"Jpn J Sarcoidosis Other Granulomatous Disord"},{"key":"2025011615465481800_R23","first-page":"1906","article-title":"[L\u00f6fgren\u2019s syndrome following breast cancer treatment] Nyugan tiryogo ni hassyo sita L\u00f6fgren syokogun","volume":"54","author":"Urano","year":"2012","journal-title":"Jpn J Clin Dermatol"},{"key":"2025011615465481800_R24","doi-asserted-by":"crossref","first-page":"91","DOI":"10.7878\/jjsogd.33.91","article-title":"Two Cases of L\u00f6fgren\u2019s syndrome: a literature-based comparison between Japanese and European clinical characteristics","volume":"33","author":"Mori","year":"2013","journal-title":"Jpn J Sarcoidosis Other Granulomatous Disord"},{"key":"2025011615465481800_R25","first-page":"578","article-title":"A case of L\u00f6fgren\u2019s syndrome with acute dyspnea","volume":"2","author":"Noda","year":"2013","journal-title":"JRS"},{"key":"2025011615465481800_R26","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1620\/tjem.234.137","article-title":"A Japanese patient with 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A retrospective survey of 49 patients with review of the literature","volume":"25","author":"Gran","year":"1996","journal-title":"Scand J Rheumatol"},{"key":"2025011615465481800_R37","doi-asserted-by":"crossref","first-page":"456","DOI":"10.1111\/j.1440-1843.2006.00874.x","article-title":"Analysis of 87 patients with Lofgren\u2019s syndrome and the pattern of seasonality of subacute sarcoidosis","volume":"11","author":"Sipahi Demirkok","year":"2006","journal-title":"Respirology"},{"key":"2025011615465481800_R38","first-page":"1622","article-title":"Osteoarticular involvement in a series of 100 patients with sarcoidosis referred to rheumatology departments","volume":"35","author":"Thelier","year":"2008","journal-title":"J Rheumatol"},{"key":"2025011615465481800_R39","doi-asserted-by":"crossref","DOI":"10.1186\/1465-9921-11-25","article-title":"Different HLA-DRB1 allele distributions in distinct clinical subgroups of sarcoidosis patients","volume":"11","author":"Grunewald","year":"2010","journal-title":"Respir Res"},{"key":"2025011615465481800_R40","first-page":"688","article-title":"Role of HLA-DR antigens in Japanese patients with sarcoidosis","volume":"135","author":"Kunikane","year":"1987","journal-title":"Am Rev Respir Dis"},{"key":"2025011615465481800_R41","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1164\/rccm.200807-1082OC","article-title":"Lofgren\u2019s syndrome: human leukocyte antigen strongly influences the disease course","volume":"179","author":"Grunewald","year":"2009","journal-title":"Am J Respir Crit Care Med"},{"key":"2025011615465481800_R42","doi-asserted-by":"crossref","first-page":"463","DOI":"10.1055\/s-0037-1602380","article-title":"Lofgren\u2019s Syndrome: diagnosis, management, and disease pathogenesis","volume":"38","author":"Karakaya","year":"2017","journal-title":"Semin Resp Crit Care Med"},{"key":"2025011615465481800_R43","doi-asserted-by":"crossref","first-page":"484","DOI":"10.1183\/13993003.00092-2016","article-title":"Expanded lung T-bet+ROR\u03b3T+ CD4+ T-cells in sarcoidosis patients with a favourable disease phenotype","volume":"48","author":"Kaiser","year":"2016","journal-title":"Eur Respir J"}],"container-title":["Modern Rheumatology Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/mrcr\/advance-article-pdf\/doi\/10.1093\/mrcr\/rxae034\/58820041\/rxae034.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/9\/1\/193\/58820041\/rxae034.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/9\/1\/193\/58820041\/rxae034.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,1,16]],"date-time":"2025-01-16T15:53:53Z","timestamp":1737042833000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/mrcr\/article\/9\/1\/193\/7724630"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7,31]]},"references-count":43,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,7,31]]},"published-print":{"date-parts":[[2025,1,16]]}},"URL":"https:\/\/doi.org\/10.1093\/mrcr\/rxae034","relation":{},"ISSN":["2472-5625"],"issn-type":[{"value":"2472-5625","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2025,1]]},"published":{"date-parts":[[2024,7,31]]}}}