{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,9]],"date-time":"2026-06-09T17:52:41Z","timestamp":1781027561388,"version":"3.54.1"},"reference-count":28,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,10,4]],"date-time":"2021-10-04T00:00:00Z","timestamp":1633305600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2021,10,4]],"date-time":"2021-10-04T00:00:00Z","timestamp":1633305600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"Medizinische Hochschule Hannover (MHH)"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Med Case Reports"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Melkersson\u2013Rosenthal syndrome is a rare disease characterized by the triad of recurrent orofacial swelling with facial paralysis and fissured dorsal tongue. Histologically, noncaseating granulomatous inflammation occurs that confirms the diagnosis. Overlaps between granulomatous diseases such as sarcoidosis and Crohn\u2019s disease are described. Systemic corticosteroid therapy is the treatment of choice for acute attacks.\n<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>We here present a case of a 59-year-old White woman suffering from Melkersson\u2013Rosenthal syndrome with a past history of sarcoidosis on therapy with leflunomide in combination with low-dose tacrolimus successfully treated with the anti-leprosy drug clofazimine after failure of systemic steroid therapy.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>We propose clofazimine as an alternative treatment in steroid-refractory cases.\n<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s13256-021-03044-5","type":"journal-article","created":{"date-parts":[[2021,10,4]],"date-time":"2021-10-04T16:57:00Z","timestamp":1633366620000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Melkersson\u2013Rosenthal syndrome in the context of sarcoidosis: a case report\u00a0"],"prefix":"10.1186","volume":"15","author":[{"given":"J.","family":"Casper","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Mohammad-Khani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. J.","family":"Schmidt","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. T.","family":"Kielstein","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T.","family":"Lenarz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H.","family":"Haller","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annette D.","family":"Wagner","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,10,4]]},"reference":[{"key":"3044_CR1","first-page":"737","volume":"90","author":"E Melkersson","year":"1928","unstructured":"Melkersson E. Case of recurrent facial paralysis with angioneurotic edema. Hygiea. 1928;90:737\u201341.","journal-title":"Hygiea"},{"key":"3044_CR2","doi-asserted-by":"publisher","first-page":"475","DOI":"10.1007\/BF02865984","volume":"131","author":"C Rosenthal","year":"1931","unstructured":"Rosenthal C. Klinisch-erbbiologischer Beitrag zur Konstitutionspathologie. Gemeinsames Auftreten von Facialisl\u00e4hmung, angioneurotischem Gesichts\u00f6dem und Lingua plicata in Arthritismus-Familien. Z Gesamte Neurol Psychiatr. 1931;131:475\u2013501.","journal-title":"Z Gesamte Neurol Psychiatr"},{"issue":"12","key":"3044_CR3","first-page":"1557","volume":"136","author":"RM Greene","year":"2000","unstructured":"Greene RM, Rogers RS. Melkersson-Rosenthal syndrome and orofacial granulomatosis. Arch Dermatol. 2000;136(12):1557\u20138.","journal-title":"Arch Dermatol"},{"key":"3044_CR4","doi-asserted-by":"crossref","unstructured":"Greene RM, Rogers RS. Melkersson-Rosenthal syndrome: a review of 36 patients. 1989;21(6):1263\u201370.","DOI":"10.1016\/S0190-9622(89)70341-8"},{"issue":"4","key":"3044_CR5","doi-asserted-by":"publisher","first-page":"191","DOI":"10.1111\/j.1600-0714.2007.00591.x","volume":"37","author":"WM Tilakaratne","year":"2008","unstructured":"Tilakaratne WM, Freysdottir J, Fortune F. Orofacial granulomatosis: review on aetiology and pathogenesis. Oral Pathol Med. 2008;37(4):191\u20135.","journal-title":"Oral Pathol Med"},{"issue":"10","key":"3044_CR6","doi-asserted-by":"publisher","first-page":"576","DOI":"10.1034\/j.1600-0714.2003.t01-1-00056.x","volume":"32","author":"JJ Sciubba","year":"2003","unstructured":"Sciubba JJ, Said-Al-Naief N. Orofacial granulomatosis: presentation, pathology and management of 13 cases. 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Int J Lepr Other Mycobact Dis. 1987;55(1):9\u201315.","journal-title":"Int J Lepr Other Mycobact Dis"}],"container-title":["Journal of Medical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-021-03044-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s13256-021-03044-5\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-021-03044-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,10,4]],"date-time":"2021-10-04T17:00:16Z","timestamp":1633366816000},"score":1,"resource":{"primary":{"URL":"https:\/\/jmedicalcasereports.biomedcentral.com\/articles\/10.1186\/s13256-021-03044-5"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,10,4]]},"references-count":28,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["3044"],"URL":"https:\/\/doi.org\/10.1186\/s13256-021-03044-5","relation":{},"ISSN":["1752-1947"],"issn-type":[{"value":"1752-1947","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,10,4]]},"assertion":[{"value":"17 February 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"3 August 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"4 October 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Ethical approval was obtained, and the patient gave written, informed consent to publish this case and any accompanying images.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"488"}}