{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T16:34:11Z","timestamp":1791995651600,"version":"4.4.0"},"reference-count":54,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2020,1,29]],"date-time":"2020-01-29T00:00:00Z","timestamp":1580256000000},"content-version":"vor","delay-in-days":28,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100003478","name":"Ministry of Health, Labour and Welfare","doi-asserted-by":"publisher","award":["Health and Labour Sciences Research Grants for Res"],"award-info":[{"award-number":["Health and Labour Sciences Research Grants for Res"]}],"id":[{"id":"10.13039\/501100003478","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatrics International"],"published-print":{"date-parts":[[2020,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Fibrodysplasia ossificans progressiva (FOP) is a rare genetic skeletal disorder manifesting progressive heterotopic ossification (HO) and congenital malformation of the great toes. Since 2007, we have conducted research on FOP. Here, we review the findings on FOP published to date, including the results of our research. Epidemiological studies in Japan have indicated that FOP has nearly the same prevalence in Japan as in the rest of the world. Basic research on its pathoetiology has progressed rapidly since the identification of the causal gene in 2006. Clinical and radiological findings have been thoroughly researched, including early radiological signs, and diagnostic criteria were established, designating FOP as an intractable disease in Japan. In patients with FOP, the progression of HO is associated with numerous disabilities, often manifesting in vicious cycles that can lead to early mortality. Through cross\u2010sectional and short\u2010term longitudinal studies, we have explored patient education, quality of life, and activities of daily living among Japanese patients. The management of FOP requires education of patients and caregivers, the use of medications to settle inflammation and flare\u2010ups, instructions to ensure proper oral care, and other compensatory approaches that aid in rehabilitation. An avoidance of medical intervention, which may cause HO to progress, is also important. The advent of new drugs to prevent HO could have clinical benefit.<\/jats:p>","DOI":"10.1111\/ped.14065","type":"journal-article","created":{"date-parts":[[2019,11,27]],"date-time":"2019-11-27T15:27:52Z","timestamp":1574868472000},"page":"3-13","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Fibrodysplasia ossificans progressiva: Review and research activities in Japan"],"prefix":"10.1111","volume":"62","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7806-7641","authenticated-orcid":false,"given":"Nobuhiko","family":"Haga","sequence":"first","affiliation":[{"name":"Department of Rehabilitation Medicine The University of Tokyo Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasuharu","family":"Nakashima","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery Kyushu University Fukuoka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Kitoh","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery Aichi Children\u2019s Health and Medical Center Obu Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Junji","family":"Kamizono","sequence":"additional","affiliation":[{"name":"Emergency Care Services Children\u2019s Medical Center Kitakyushu City YAHATA Hospital Kitakyushu Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9304-2098","authenticated-orcid":false,"given":"Takenobu","family":"Katagiri","sequence":"additional","affiliation":[{"name":"Division of Pathophysiology Research Center for Genomic Medicine Saitama Medical University Saitama Japan"},{"name":"Project of Clinical and Basic Research for FOP Saitama Medical University Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hideto","family":"Saijo","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery The University of Tokyo Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sho","family":"Tsukamoto","sequence":"additional","affiliation":[{"name":"Division of Pathophysiology Research Center for Genomic Medicine Saitama Medical University Saitama Japan"},{"name":"Project of Clinical and Basic Research for FOP Saitama Medical University Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5489-2751","authenticated-orcid":false,"given":"Yusuke","family":"Shinoda","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Medicine The University of Tokyo Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryoko","family":"Sawada","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Medicine The University of Tokyo Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasuo","family":"Nakahara","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Teikyo University Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,1,29]]},"reference":[{"key":"e_1_2_11_2_1","unstructured":"Research Group on fibrodysplasia ossificans progressiva. 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