{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,2]],"date-time":"2025-08-02T00:11:37Z","timestamp":1754093497047,"version":"3.41.2"},"reference-count":22,"publisher":"Oxford University Press (OUP)","issue":"8","license":[{"start":{"date-parts":[[2025,6,18]],"date-time":"2025-06-18T00:00:00Z","timestamp":1750204800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"name":"Radiant Hope Foundation"},{"name":"Robert and Arlene Kogod Professorship in Geriatric Medicine"},{"DOI":"10.13039\/100030709","name":"Center for Research in FOP and Related Disorders","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100030709","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Ian Cali Endowment for FOP Research"},{"name":"Isaac and Rose Nassau Professorship of Orthopedic Molecular Medicine"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,7,5]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Pseudoarthroses (false joints) occur as a complication of fracture healing in the normotopic skeleton but have not been reported at ectopic sites. Fibrodysplasia ossificans progressiva (FOP) is an ultra-rare genetic disorder characterized by progressive heterotopic ossification (HO) and congenital skeletal abnormalities including developmental arthropathy in the normotopic skeleton. Here, we report ectopic pseudojoint formation in 6 patients with FOP, all occurring after painful flare-ups and HO in soft connective tissues of the knee, hip, and forearm. To our knowledge, FOP is the only human condition in which ectopic pseudojoint formation occurs. These findings support that dysregulated BMP pathway signaling from mutant ACVR1 mediates not only HO, but also ectopic pseudojoint formation, and that ectopic pseudojoints can arise from de novo musculoskeletal elements.<\/jats:p>","DOI":"10.1093\/jbmrpl\/ziaf107","type":"journal-article","created":{"date-parts":[[2025,6,27]],"date-time":"2025-06-27T11:12:22Z","timestamp":1751022742000},"source":"Crossref","is-referenced-by-count":0,"title":["Ectopic pseudojoints in fibrodysplasia ossificans progressiva"],"prefix":"10.1093","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8533-9438","authenticated-orcid":false,"given":"Robert J","family":"Pignolo","sequence":"first","affiliation":[{"name":"Department of Medicine, Mayo Clinic College of Medicine, Mayo Clinic , Rochester, MN 55905 ,","place":["United States"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7138-596X","authenticated-orcid":false,"given":"Mona","family":"Al Mukaddam","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]},{"name":"Department of Medicine, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]},{"name":"Center for Research in FOP and Related Disorders, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Stephen","family":"Broski","sequence":"additional","affiliation":[{"name":"Department of Radiology, Mayo Clinic , Rochester, MN 55905 ,","place":["United States"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rolf","family":"Morhart","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Klinikum Garmisch-Partenkirchen GmbH , Garmisch-Partenkirchen, 82467 ,","place":["Germany"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Luke T","family":"Drake","sequence":"additional","affiliation":[{"name":"Department of Medicine, Mayo Clinic College of Medicine, Mayo Clinic , Rochester, MN 55905 ,","place":["United States"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bruce","family":"Kneeland","sequence":"additional","affiliation":[{"name":"Department of Radiology, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"O Will","family":"Towler","sequence":"additional","affiliation":[{"name":"Center for Research in FOP and Related Disorders, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]},{"name":"Division of Plastic Surgery, Department of Surgery, Children\u2019s Hospital of Philadelphia , Philadelphia, PA 19104 ,","place":["United States"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Frederick S","family":"Kaplan","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]},{"name":"Department of Medicine, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]},{"name":"Center for Research in FOP and Related Disorders, Perelman School of Medicine at the University of Pennsylvania , Philadelphia, PA 19104 ,","place":["United States"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2025,6,18]]},"reference":[{"issue":"1","key":"2025080110272094300_ref1","doi-asserted-by":"publisher","first-page":"350","DOI":"10.1186\/s13023-021-01983-2","article-title":"Prevalence of fibrodysplasia ossificans progressiva (FOP) in the United 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