{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T07:55:24Z","timestamp":1791446124929,"version":"4.3.3"},"reference-count":8,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T00:00:00Z","timestamp":1784592000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T00:00:00Z","timestamp":1784592000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The Journal of Dermatology"],"published-print":{"date-parts":[[2026,10]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Multicentric reticulohistiocytosis (MRH) is a rare non\u2010Langerhans cell histiocytosis characterized by destructive polyarthritis and papulonodular cutaneous lesions. We describe a woman who developed erythroderma 20\u2009years after MRH diagnosis\u2014to our knowledge, a previously unreported manifestation of this disease. The patient initially presented at age 50 with arthralgia, and MRH was diagnosed one year later based on distal interphalangeal (DIP) joint deformities, erythematous papules on the lower legs, and a skin biopsy showing dermal infiltration of CD68\u2010positive, S\u2010100\u2013 and CD1a\u2013negative histiocyte\u2010like cells with ground\u2010glass eosinophilic cytoplasm. Despite long\u2010term treatment with methotrexate (MTX), prednisolone (PSL), infliximab (year 11), and bone\u2010modifying agents (alendronate, later denosumab), the disease progressed, with development of pulmonary arterial hypertension (PAH) attributable to lesions around the upper pulmonary veins. At age 71, twenty years after diagnosis, pruritic erythema appeared on the back and evolved into erythroderma despite topical corticosteroids. Biopsies from erythematous areas demonstrated CD68\u2010positive histiocytic infiltrate identical to that of the original MRH lesions, whereas biopsies from clinically uninvolved skin showed no such infiltration, supporting a direct association with the MRH disease process. Comprehensive evaluation excluded eczematous\/atopic erythroderma (normal eosinophils and IgE), drug\u2010induced erythroderma (no new medications; infliximab discontinued 9\u2009years previously), dermatophytosis (negative PAS staining), granulomatous mycosis fungoides (absent characteristic histological features), sarcoidosis (normal sIL\u20102R; no granulomas on biopsy), dermatomyositis (absent characteristic features; normal creatine kinase), and internal malignancy (contrast\u2010enhanced CT and 2\u2010year follow\u2010up). MTX was discontinued because of MTX\u2010induced interstitial pneumonia, and oral PSL was increased to 30\u2009mg\/day, with rapid improvement; the patient is currently maintained on PSL 10\u2009mg\/day without relapse. The cumulative inflammatory burden of long\u2010standing MRH may have culminated in this erythrodermic phenotype.<\/jats:p>","DOI":"10.1111\/1346-8138.70415","type":"journal-article","created":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T08:36:13Z","timestamp":1784622973000},"page":"1542-1546","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Multicentric Reticulohistiocytosis Progressing to Erythroderma After Long\u2010Term Disease Progression"],"prefix":"10.1111","volume":"53","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2519-8378","authenticated-orcid":false,"given":"Ayano","family":"Maruyama","sequence":"first","affiliation":[{"name":"Department of Dermatology, Graduate School of Medical Science Kyoto Prefectural University of Medicine  Kyoto Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Koji","family":"Masuda","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Graduate School of Medical Science Kyoto Prefectural University of Medicine  Kyoto Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0364-711X","authenticated-orcid":false,"given":"Takeshi","family":"Fukumoto","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Graduate School of Medical Science Kyoto Prefectural University of Medicine  Kyoto Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,7,21]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1001\/archderm.1954.01540180067010"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/kez555"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11926-015-0511-6"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.berh.2004.06.002"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.ASW.0000463573.40637.73"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2020.12.010"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1001\/archdermatol.2008.46"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamadermatol.2021.0996"}],"container-title":["The Journal of Dermatology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/1346-8138.70415","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/1346-8138.70415","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/1346-8138.70415","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T07:35:20Z","timestamp":1791444920000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/1346-8138.70415"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,7,21]]},"references-count":8,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2026,10]]}},"alternative-id":["10.1111\/1346-8138.70415"],"URL":"https:\/\/doi.org\/10.1111\/1346-8138.70415","archive":["Portico"],"relation":{},"ISSN":["0385-2407","1346-8138"],"issn-type":[{"value":"0385-2407","type":"print"},{"value":"1346-8138","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,7,21]]},"assertion":[{"value":"2026-04-15","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-16","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-21","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}