{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,13]],"date-time":"2026-09-13T22:54:16Z","timestamp":1789340056998,"version":"build-2803163510"},"reference-count":11,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,2,26]],"date-time":"2021-02-26T00:00:00Z","timestamp":1614297600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,2,26]],"date-time":"2021-02-26T00:00:00Z","timestamp":1614297600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Rheumatol"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Introduction<\/jats:title>\n                <jats:p>Calcinosis cutis is a common complication of pediatric rheumatologic diseases. However, there is currently no consensus on first-line treatment. Bisphosphonates have been described as a successful treatment in several case studies, but most of these cases are limited to patients with isolated juvenile dermatomyositis or systemic sclerosis. Specifically, there are limited reports of their usefulness in treating overlap syndromes and mixed connective tissue disorders.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>We describe the case of a 13\u2009year-old girl with overlap syndrome with features of juvenile dermatomyositis and systemic lupus erythematosus. After 22\u2009months of extensive immunosuppressive therapy, including monthly IVIG and Rituximab, she continued to have pain and weakness of the lower extremities. A CT scan was performed which showed significant multifocal soft tissue calcifications of the pelvis. She was started on treatment with oral alendronate with the goal of improving her calcinosis and improving her symptoms. After several months of therapy, our patient reported subjective improvement of her lower extremity pain and weakness, as well as complete resolution of abnormalities previously seen on physical examination. A repeat CT scan of the pelvis was performed after 11\u2009months of therapy and demonstrated complete resolution of the previously seen calcinosis.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>We report the successful treatment of soft tissue calcinosis with oral bisphosphonates in a patient with juvenile dermatomyositis-systemic lupus erythematosus overlap syndrome. These results provide further evidence that bisphosphonates can be used successfully to treat calcinosis cutis in pediatric rheumatologic disorders. Additionally, the results provide new evidence that they can be used specifically in juvenile dermatomyositis-systemic lupus erythematosus overlap syndrome, which has not been previously reported.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s41927-021-00176-5","type":"journal-article","created":{"date-parts":[[2021,2,26]],"date-time":"2021-02-26T00:05:09Z","timestamp":1614297909000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Resolution of calcinosis using bisphosphonates in overlap syndrome \u2013 a case report"],"prefix":"10.1186","volume":"5","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3983-956X","authenticated-orcid":false,"given":"Mitchell","family":"Platter","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brian","family":"Pugmire","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Reshma","family":"Patel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,2,26]]},"reference":[{"key":"176_CR1","doi-asserted-by":"publisher","unstructured":"Mathiesen P, Hegaard H, Herlin T, Zak M, Pedersen FK, Nielsen S. Long-term outcome in patients with juvenile dermatomyositis: a cross-sectional follow-up study. Scand J Rheumatol. 2012. https:\/\/doi.org\/10.3109\/03009742.2011.608376.","DOI":"10.3109\/03009742.2011.608376"},{"issue":"4","key":"176_CR2","doi-asserted-by":"publisher","first-page":"549","DOI":"10.1590\/S0482-50042012000400007","volume":"52","author":"G Clemente","year":"2012","unstructured":"Clemente G, Piotto DG, Barbosa C, Peracchi OA, Len CA, Hilario MO, et al. High frequency of calcinosis in juvenile dermatomyositis: a risk factor study. Rev Bras Rheumatol. 2012;52(4):549\u201353.","journal-title":"Rev Bras Rheumatol"},{"key":"176_CR3","unstructured":"Gara S, Jamil RT, Litaiem N. Juvenile dermatomyositis. StatPearls StatPearls Publishing https:\/\/www.ncbi.nlm.nih.gov\/books\/NBK534236. Accessed 31 Mar 2020."},{"key":"176_CR4","doi-asserted-by":"publisher","unstructured":"Balin SJ, Wetter DA, Andersen LK, Davis MDP. Calcinosis cutis occurring in association with autoimmune connective tissue disease: the Mayo Clinic experience with 78 patients, 1996-2009. Arch Dermatol. 2012. https:\/\/doi.org\/10.1001\/archdermatol.2011.2052.","DOI":"10.1001\/archdermatol.2011.2052"},{"issue":"3","key":"176_CR5","first-page":"538","volume":"43","author":"T Miyamae","year":"2003","unstructured":"Miyamae T, Mori M, Inamo Y, Kohno Y, Takei S, Maeda M, et al. Multi-center analysis of calcinosis in children with juvenile dermatomyositis. Ryumachi. 2003;43(3):538\u201343.","journal-title":"Ryumachi."},{"key":"176_CR6","doi-asserted-by":"publisher","unstructured":"Orandi AB, Baszis KW, Dharnidharka VR, Huber AM, Hoeltzel MF; CARRA Juvenile Myositis subgroup. Assessment, classification and treatment of calcinosis as a complication of juvenile dermatomyositis: a survey of pediatric rheumatologists by the childhood arthritis and rheumatology research alliance (CARRA). Pediatr Rheumatol Online J. 2017; doi:https:\/\/doi.org\/10.1186\/s12969-017-0199-4.","DOI":"10.1186\/s12969-017-0199-4"},{"key":"176_CR7","doi-asserted-by":"publisher","unstructured":"Tayfur AC, Topaloglu R, Gulhan B, Bilginer Y. Bisphosphonates in juvenile dermatomyositis with dystrophic calcinosis. Mod Rheumatol. 2015. https:\/\/doi.org\/10.3109\/14397595.2014.988197.","DOI":"10.3109\/14397595.2014.988197"},{"key":"176_CR8","doi-asserted-by":"publisher","unstructured":"Martillotti J, Moote D, Zemel L. Improvement of calcinosis using pamidronate in a patient with juvenile dermatomyositis. Pediatr Radiol. 2014. https:\/\/doi.org\/10.1007\/s00247-013-2738-6.","DOI":"10.1007\/s00247-013-2738-6"},{"issue":"2","key":"176_CR9","doi-asserted-by":"publisher","first-page":"88","DOI":"10.1046\/j.1440-1754.2003.00083.x","volume":"39","author":"JA Batch","year":"2003","unstructured":"Batch JA, Couper JJ, Rodda C, Cowell CT, Zacharin M. Use of bisphosphonate therapy for osteoporosis in childhood and adolescence. J Pediatr Child Health. 2003;39(2):88\u201392.","journal-title":"J Pediatr Child Health"},{"issue":"2","key":"176_CR10","doi-asserted-by":"publisher","first-page":"219","DOI":"10.1002\/acr.20071","volume":"62","author":"AM Huber","year":"2010","unstructured":"Huber AM, Giannini EH, Bowyer SL, et al. Protocols for the initial treatment of moderately severe juvenile dermatomyositis: results of a Children's arthritis and rheumatology research Alliance consensus conference. Arthritis Care Res (Hoboken). 2010;62(2):219\u201325. https:\/\/doi.org\/10.1002\/acr.20071.","journal-title":"Arthritis Care Res (Hoboken)"},{"issue":"1","key":"176_CR11","doi-asserted-by":"publisher","first-page":"80","DOI":"10.1210\/edrv.19.1.0325","volume":"19","author":"H Fleisch","year":"1998","unstructured":"Fleisch H. Bisphosphonates: mechanisms of action. Endocr Rev. 1998;19(1):80\u2013100.","journal-title":"Endocr Rev"}],"container-title":["BMC Rheumatology"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s41927-021-00176-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s41927-021-00176-5\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s41927-021-00176-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,2,26]],"date-time":"2021-02-26T00:40:34Z","timestamp":1614300034000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcrheumatol.biomedcentral.com\/articles\/10.1186\/s41927-021-00176-5"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,2,26]]},"references-count":11,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["176"],"URL":"https:\/\/doi.org\/10.1186\/s41927-021-00176-5","relation":{},"ISSN":["2520-1026"],"issn-type":[{"value":"2520-1026","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,2,26]]},"assertion":[{"value":"11 September 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"20 January 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"26 February 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Written and verbal consent obtained from patient and parent (legal guardian).","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written consent to publish the above information, including all images, was obtained from the patient\u2019s parent, who is her legal guardian.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"6"}}