{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,22]],"date-time":"2026-09-22T11:03:29Z","timestamp":1790075009238,"version":"4.0.1"},"reference-count":59,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"5","funder":[{"name":"None","award":["None"],"award-info":[{"award-number":["None"]}]}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:p>\n                    <jats:list list-type=\"plain\">\n                      <jats:list-item>\n                        <jats:p>\u00bb Fibrodysplasia ossificans progressiva (FOP), the most severe form of heterotopic ossification (HO) in humans, may lead to severe thoracic deformity (TD) and thoracic insufficiency syndrome (TIS), the most common cause of mortality in individuals with FOP.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>\u00bb In this study, we examine the multiple causes of TD in FOP and propose a dynamic model for the development and evolution of TD that can be used to guide clinical care. This model posits that multiple factors, all originating from the causative gain-of-function mutation in activin receptor A, type 1, lead to TD in FOP.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>\u00bb Factors that lead to TD in FOP include early developmental costovertebral and facet joint arthropathy and joint ankylosis, progressive episodic flare-ups (both trauma induced or spontaneous) and subsequent HO, dysregulated soft tissue sensitivity to mechanical stimuli, growth plate dysregulation, and congenital rib fusions. These factors contribute to TD in FOP through a combination of intra-articular and extra-articular mechanisms, all amplified by growth.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>\u00bb Although FOP is a rare condition, it is illustrative of how a mutation in a critical receptor in the bone morphogenetic protein signaling pathway can cause a litany of musculoskeletal dysfunction that can lead to life-threatening consequences. Clinicians caring for individuals who have FOP must be aware of the occurrence, evolution, and implications of progressive TD and resultant TIS so that appropriate preventive measures such as avoidance of pulmonary infections, incentive spirometry, and diaphragmatic strengthening can be instituted.<\/jats:p>\n                      <\/jats:list-item>\n                    <\/jats:list>\n                  <\/jats:p>","DOI":"10.2106\/jbjs.rvw.25.00042","type":"journal-article","created":{"date-parts":[[2025,5,22]],"date-time":"2025-05-22T14:07:26Z","timestamp":1747922846000},"update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":6,"title":["Thoracic Deformity in Fibrodysplasia Ossificans Progressiva"],"prefix":"10.2106","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1277-5420","authenticated-orcid":false,"given":"Frederick S.","family":"Kaplan","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania"},{"name":"Department of Medicine, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania"},{"name":"Center for Research in FOP and Related Disorders, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2934-6374","authenticated-orcid":false,"given":"Paul D.","family":"Sponseller","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, The Johns Hopkins Hospital, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8533-9438","authenticated-orcid":false,"given":"Robert J.","family":"Pignolo","sequence":"additional","affiliation":[{"name":"Department of Medicine, Mayo Clinic College of Medicine, Mayo Clinic, Rochester, Minnesota"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2025,5]]},"reference":[{"issue":"10","key":"r1-20250522","doi-asserted-by":"crossref","first-page":"1442","DOI":"10.2106\/00004623-199410000-00002","article-title":"Spinal deformity in patients who have fibrodysplasia ossificans progressiva","volume":"76","author":"Shah","year":"1994","journal-title":"J Bone Joint Surg Am"},{"issue":"5","key":"r2-20250522","doi-asserted-by":"crossref","first-page":"201","DOI":"10.1159\/000365770","article-title":"Fibrodysplasia ossificans progressiva: clinical course, genetic mutations and genotype-phenotype correlation","volume":"5","author":"H\u00fcning","year":"2014","journal-title":"Mol Syndromol"},{"issue":"2","key":"r3-20250522","doi-asserted-by":"crossref","first-page":"220","DOI":"10.2106\/00004623-199302000-00009","article-title":"The histopathology of fibrodysplasia ossificans progressiva. 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