{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,1]],"date-time":"2025-11-01T13:50:38Z","timestamp":1762005038608,"version":"3.37.3"},"reference-count":26,"publisher":"Springer Science and Business Media LLC","issue":"3","license":[{"start":{"date-parts":[[2018,10,20]],"date-time":"2018-10-20T00:00:00Z","timestamp":1539993600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/www.springer.com\/tdm"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Rheumatol Int"],"published-print":{"date-parts":[[2019,3]]},"DOI":"10.1007\/s00296-018-4179-x","type":"journal-article","created":{"date-parts":[[2018,10,20]],"date-time":"2018-10-20T13:57:25Z","timestamp":1540043845000},"page":"569-576","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Challenges in the treatment of fibrodysplasia ossificans progressiva"],"prefix":"10.1007","volume":"39","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3639-879X","authenticated-orcid":false,"given":"Kardelen","family":"Gencer-Atalay","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0987-0876","authenticated-orcid":false,"given":"Ekim Can","family":"Ozturk","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5988-7369","authenticated-orcid":false,"given":"Ilker","family":"Yagci","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6688-2347","authenticated-orcid":false,"given":"Pinar","family":"Ata","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8366-1642","authenticated-orcid":false,"given":"Kenan","family":"Delil","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0307-6890","authenticated-orcid":false,"given":"Zerrin","family":"Ozgen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3724-571X","authenticated-orcid":false,"given":"Gulseren","family":"Akyuz","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2018,10,20]]},"reference":[{"issue":"4","key":"4179_CR1","doi-asserted-by":"publisher","first-page":"242","DOI":"10.5582\/irdr.2017.01055","volume":"6","author":"Q Zijuan","year":"2017","unstructured":"Zijuan Q, Luan J, Zhou X, Cui Y, Han J (2017) Fibrodysplasia ossificans progressiva: basic understanding and experimental metods. Intractable Rare Dis Res 6(4):242\u2013248","journal-title":"Intractable Rare Dis Res"},{"key":"4179_CR2","doi-asserted-by":"publisher","first-page":"213","DOI":"10.1385\/BMM:3:3-4:213","volume":"3","author":"FS Kaplan","year":"2005","unstructured":"Kaplan FS, Glaser DL (2005) Thoracic insufficiency syndrome in patients with fibrodysplasia ossificans progressiva. Clin Rev Bone Miner Metab 3:213\u2013216","journal-title":"Clin Rev Bone Miner Metab"},{"key":"4179_CR3","doi-asserted-by":"publisher","first-page":"e654","DOI":"10.1542\/peds.2005-0469","volume":"16","author":"JA Kitterman","year":"2005","unstructured":"Kitterman JA, Kantanie S, Rocke DM, Kaplan FS (2005) Iatrogenic harm caused by diagnostic errors in fibrodysplasia ossificans progressiva. Pediatrics 16:e654\u2013e661. https:\/\/doi.org\/10.1542\/peds.2005-0469","journal-title":"Pediatrics"},{"key":"4179_CR4","doi-asserted-by":"publisher","first-page":"80","DOI":"10.1186\/1750-1172-6-80","volume":"6","author":"RJ Pignolo","year":"2011","unstructured":"Pignolo RJ, Shore EM, Kaplan FS (2011) Fibrodysplasia ossificans progressiva: clinical and genetic aspects. Orphanet J Rare Dis 6:80. https:\/\/doi.org\/10.1186\/1750-1172-6-80","journal-title":"Orphanet J Rare Dis"},{"issue":"0 2","key":"4179_CR5","first-page":"437","volume":"10","author":"RJ Pignolo","year":"2013","unstructured":"Pignolo RJ, Shore EM, Kaplan FS (2013) Fibrodysplasia ossificans progressiva: diagnosis, management, and therapeutic Horizons. Pediatr Endocrinol Rev 10(0 2):437\u2013448","journal-title":"Pediatr Endocrinol Rev"},{"issue":"2","key":"4179_CR6","first-page":"1","volume":"1","author":"FS Kaplan","year":"2003","unstructured":"Kaplan FS, Shore EM, Glaser DL, Emerson S (2003) The medical management of fibrodysplasia ossificans progressiva: Current treatment considerations. Clin Proc Intl Clin Consort FOP 1(2):1\u201372","journal-title":"Clin Proc Intl Clin Consort FOP"},{"issue":"2","key":"4179_CR7","first-page":"611","volume":"35","author":"Z Soldic","year":"2011","unstructured":"Soldic Z, Murgic J, Radic J, Dabelic N, Jazvic M, Brozic JM el al (2011) Radiation therapy in treatment of fibrodysplasia ossificans progressiva: a case report and review of the literature. Coll Antropol 35(2):611\u2013614","journal-title":"Coll Antropol"},{"key":"4179_CR8","doi-asserted-by":"publisher","first-page":"285","DOI":"10.3171\/2011.11.SPINE1164","volume":"16","author":"BT Grobelny","year":"2012","unstructured":"Grobelny BT, Rubin D, Fleischut P, Rubens E, Fogarty P, Fink M et al (2012) Neurosurgical management of symptomatic thoracic spinal ossification in a patient with fibrodysplasia ossificans progressiva. J Neurosyrg Spine 16:285\u2013288","journal-title":"J Neurosyrg Spine"},{"issue":"8","key":"4179_CR9","doi-asserted-by":"publisher","first-page":"588","DOI":"10.7326\/0003-4819-158-8-201304160-00003","volume":"158","author":"PJ Pickhardt","year":"2013","unstructured":"Pickhardt PJ, Pooler BD, Lauder T, Munoz del Rio A, Bruce RJ, Binkley N (2013) Opportunistic screening for osteoporosis using abdominal computed tomography scans obtained for other indications. Ann Intern Med 158(8):588\u2013595","journal-title":"Ann Intern Med"},{"issue":"4","key":"4179_CR10","first-page":"4454","volume":"6","author":"O Rogoveanu","year":"2013","unstructured":"Rogoveanu O, Traistrau R, Streba CT, Stoica Z, Popescu R (2013) Clinical, evolution and therapeutical considerations upon a case of fibrodysplasia ossificans progressiva (FOP). J Med Life 6(4):454\u2013458","journal-title":"J Med Life"},{"key":"4179_CR11","first-page":"6","volume":"271","author":"B Bouvard","year":"2016","unstructured":"Bouvard B, Masson C, Legrand E, Audran M (2016) Fibrodyspalsia ossificans progressiva. A case report and focus on the BMP signaling pathway. Morpho 271:6","journal-title":"Morpho"},{"issue":"5","key":"4179_CR12","doi-asserted-by":"publisher","first-page":"1379","DOI":"10.1007\/s00296-010-1426-1","volume":"32","author":"H Ulusoy","year":"2012","unstructured":"Ulusoy H (2012) Fibrodysplasia ossificans progressiva without characteristic skeletal anomalies. Rheumatol Int 32(5):1379\u20131382. https:\/\/doi.org\/10.1007\/s00296-010-1426-1","journal-title":"Rheumatol Int"},{"issue":"6","key":"4179_CR13","doi-asserted-by":"crossref","first-page":"1460","DOI":"10.1359\/jbmr.091113","volume":"25","author":"D Gatti","year":"2010","unstructured":"Gatti D, Viapiana O, Rossini M, Silvano A (2010) Rosiglitazone therapy is assosiated with major clinical improvements in a patient with fibrodysplasia ossificans progressiva. J Bone Miner Res 25(6):1460\u20131462","journal-title":"J Bone Miner Res"},{"issue":"3","key":"4179_CR14","doi-asserted-by":"publisher","first-page":"270","DOI":"10.3109\/10582452.2010.501300","volume":"18","author":"DB Palhares","year":"2010","unstructured":"Palhares DB, Leme LM, Naka EN, Melnikov P (2010) Fibrodysplasia ossificans progressiva: oral ascorbate and intravenous bisphosphonate during flare-ups. J Musculoskelet Pain 18(3):270\u2013276","journal-title":"J Musculoskelet Pain"},{"key":"4179_CR15","doi-asserted-by":"publisher","first-page":"281","DOI":"10.1016\/j.bone.2017.12.011","volume":"109","author":"FS Kaplan","year":"2018","unstructured":"Kaplan FS, Zeitlin L, Dunn SP, Benor S, Hagin D, Mukaddam AM et al (2018) Acute and chronic rapamycin use in patients with fibrodysplasia ossificans progressiva: a report of two cases. Bone 109:281\u2013284","journal-title":"Bone"},{"key":"4179_CR16","doi-asserted-by":"publisher","first-page":"276","DOI":"10.1016\/j.bone.2017.07.019","volume":"109","author":"FS Kaplan","year":"2018","unstructured":"Kaplan FS, Andolina JR, Adamson PC, Teachey DT, Finklestein JZ, Ebb DH et al (2018) Early clinical observations on the use of imatinib mesylate in FOP: a report of seven cases. Bone 109:276\u2013280","journal-title":"Bone"},{"key":"4179_CR17","doi-asserted-by":"publisher","first-page":"163","DOI":"10.1186\/1750-1172-8-163","volume":"8","author":"H Kitoh","year":"2013","unstructured":"Kitoh H, Achiwa M, Kaneko H, Mishima K, Matsushita M, Kadono I et al (2013) Perhexiline maleate in the treatment of fibrodysplasia ossificans progressiva: an open-labeled clinical trial. Orphanet J Rare Dis 8:163","journal-title":"Orphanet J Rare Dis"},{"key":"4179_CR18","first-page":"192","volume":"62","author":"S Ozkan","year":"2014","unstructured":"Ozkan S, Alp E, Demirag F, Yazici U, Karaoglanoglu N (2014) Intrathoracic Fibrodysplasia Ossificans Progressiva. Thorac Cardiovasc Surg 62:192\u2013195","journal-title":"Thorac Cardiovasc Surg"},{"key":"4179_CR19","doi-asserted-by":"publisher","first-page":"61","DOI":"10.5761\/atcs.cr.11.01677","volume":"18","author":"Y Seok","year":"2012","unstructured":"Seok Y, Cho S, Lee E (2012) Surgical treatment combined with NSAIDs in Fibrodysplasia ossificans progressiva. Ann Thorac Cardiovasc Surg 18:61\u201363","journal-title":"Ann Thorac Cardiovasc Surg"},{"issue":"4","key":"4179_CR20","first-page":"e83","volume":"53","author":"JASB Jayasundara","year":"2012","unstructured":"Jayasundara JASB, Punchihewa GL, Alwis DS (2012) An unusual case of adult onset progressive heterotopic ossification suggesting a variant form of fibrodysplasia ossificans progressiva. Singap Med J 53(4):e83\u2013e86","journal-title":"Singap Med J"},{"key":"4179_CR21","doi-asserted-by":"publisher","first-page":"1408","DOI":"10.1016\/j.joms.2009.11.005","volume":"68","author":"Y Duan","year":"2010","unstructured":"Duan Y, Zhang H, Bu R (2010) Intraoral approach technique for treathing trismus caused by fibrodysplasia ossificans progressiva. J Oral Maxillofac Surg 68:1408\u20131410","journal-title":"J Oral Maxillofac Surg"},{"key":"4179_CR22","doi-asserted-by":"publisher","first-page":"1497","DOI":"10.2106\/JBJS.H.00554","volume":"91","author":"RE Moore","year":"2009","unstructured":"Moore RE, Dormans JP, Drummond DS, Shore EM, Kaplan FS, Auerbach JD (2009) Chin-on-chest deformity in patients with fibrodysplasia ossificans progressiva. J Bone Jt Surg Am 91:1497\u20131502","journal-title":"J Bone Jt Surg Am"},{"issue":"2","key":"4179_CR23","doi-asserted-by":"publisher","first-page":"813","DOI":"10.22271\/ortho.2017.v3.i2i.88","volume":"3","author":"N Dharra","year":"2017","unstructured":"Dharra N, Srivastava R, Halder S, Hukku S (2017) Role of radiotherapy in management of Fibrodysplasia ossificans progressiva. Int J Orthop Sci 3(2):813\u2013816","journal-title":"Int J Orthop Sci"},{"issue":"2","key":"4179_CR24","first-page":"611","volume":"35","author":"Z Soldic","year":"2011","unstructured":"Soldic Z, Murgic J, Radic J, Dabelic N, Jazvic M, Brozic JM et al (2011) Radiation therapy in treatment of fibrodysplasia ossificans progressiva: a case report and review of the literature. Coll Antropol 35(2):611\u2013614","journal-title":"Coll Antropol"},{"key":"4179_CR25","doi-asserted-by":"publisher","first-page":"989","DOI":"10.3390\/ijms19040989","volume":"19","author":"S Cappato","year":"2018","unstructured":"Cappato S, Giacopelli F, Ravazzolo R, Bocciardi R (2018) The horizon of a therapy for rare genetic diseases: a \u201cDruggable\u201d future for fibrodysplasia ossificans progressiva. Int J Mol Sci 19:989. https:\/\/doi.org\/10.3390\/ijms19040989","journal-title":"Int J Mol Sci"},{"key":"4179_CR26","doi-asserted-by":"publisher","first-page":"259","DOI":"10.1016\/j.bone.2017.08.023","volume":"109","author":"TA Brennan","year":"2018","unstructured":"Brennan TA, Lindborg CM, Bergbauer CR, Wang H, Kaplan FS, Pignolo RJ (2018) Mast cell inhibition as a therapeutic approach in fibrodysplasia ossificans progressiva (FOP). Bone 109:259\u2013266. https:\/\/doi.org\/10.1016\/j.bone.2017.08.023","journal-title":"Bone"}],"container-title":["Rheumatology International"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/article\/10.1007\/s00296-018-4179-x\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s00296-018-4179-x.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s00296-018-4179-x.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,7,11]],"date-time":"2024-07-11T16:00:08Z","timestamp":1720713608000},"score":1,"resource":{"primary":{"URL":"http:\/\/link.springer.com\/10.1007\/s00296-018-4179-x"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,10,20]]},"references-count":26,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2019,3]]}},"alternative-id":["4179"],"URL":"https:\/\/doi.org\/10.1007\/s00296-018-4179-x","relation":{},"ISSN":["0172-8172","1437-160X"],"issn-type":[{"type":"print","value":"0172-8172"},{"type":"electronic","value":"1437-160X"}],"subject":[],"published":{"date-parts":[[2018,10,20]]},"assertion":[{"value":"4 August 2018","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"15 October 2018","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"20 October 2018","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Compliance with ethical standards"}},{"value":"KGA declares that she has no conflict of interest. ECO declares that he has no conflict of interest. IY declares that he has no conflict of interest. PA declares that she has no conflict of interest. KD declares that he has no conflict of interest. ZO declares that she has no conflict of interest. GA declares that she has no conflict of interest.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and\/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical approval"}},{"value":"Informed consent was obtained from the patient included in the study.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Informed consent"}}]}}