{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,29]],"date-time":"2026-05-29T10:25:29Z","timestamp":1780050329928,"version":"3.53.1"},"reference-count":47,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2022,3,22]],"date-time":"2022-03-22T00:00:00Z","timestamp":1647907200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:p>Fibrodysplasia ossificans progressiva (FOP) is a rare genetic disorder characterized by heterotopic ossification (HO) of the skeletal muscles, fascia, tendons and ligaments. Patients often experience limitations in jaw function due to HO formation in the maxillofacial region. However, no studies have yet analyzed the age of onset and location of HO and the type of restrictions it may yield in the maxillofacial region. The aim of this study was to evaluate all existing literature on the site of onset of HO and associated functional restrictions of the jaw. To this end, a scoping review was performed focusing on limitations of jaw movement in FOP patients. The literature search resulted in 725 articles, of which 30 articles were included for full study after applying the exclusion criteria. From these articles 94 FOP patients were evaluated for gender, age, presence and age at which HO started in the maxillofacial region, location of HO, whether HO was caused spontaneous or traumatic and maximum mouth opening. Formation of HO is slightly more common in female patients compared to male patients, but the age of HO onset or the maximum mouth opening does not differ between genders. Trauma-induced HO occurred at a significantly younger age than spontaneous HO. Interestingly, a difference in maximum mouth opening was observed between the different ossified locations in the maxillofacial region, with ossification of the masseter muscle resulting in the smallest and ossification of the zygomatic arch resulting in the largest maximum mouth opening. This review revealed that the location of the maxillofacial region affected by HO determines the degree of limitations of the maximum mouth opening. This finding may be important for establishing clinical guidelines for the dental management of FOP patients.<\/jats:p>","DOI":"10.3389\/fmed.2022.852678","type":"journal-article","created":{"date-parts":[[2022,3,27]],"date-time":"2022-03-27T21:05:55Z","timestamp":1648415155000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":5,"title":["Limitations of Jaw Movement in Fibrodysplasia Ossificans Progressiva: A Review"],"prefix":"10.3389","volume":"9","author":[{"given":"Ton","family":"Schoenmaker","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amine","family":"Dahou Bouchankouk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Semih","family":"\u00d6zkan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marjolijn","family":"Gilijamse","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elinor","family":"Bouvy-Berends","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Coen","family":"Netelenbos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank","family":"Lobbezoo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisabeth Marelise W.","family":"Eekhoff","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teun J.","family":"de Vries","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2022,3,22]]},"reference":[{"key":"B1","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 States: estimate from three treatment centers and a patient organization","volume":"16","author":"Pignolo","year":"2021","journal-title":"Orphanet J Rare Dis."},{"key":"B2","doi-asserted-by":"publisher","first-page":"123","DOI":"10.1186\/s13023-017-0674-5","article-title":"Prevalence of fibrodysplasia ossificans progressiva (FOP) in France: an estimate based on a record linkage of two national databases","volume":"12","author":"Baujat","year":"2017","journal-title":"Orphanet J Rare Dis."},{"key":"B3","doi-asserted-by":"publisher","first-page":"124","DOI":"10.1016\/j.bone.2017.06.009","article-title":"Joint-specific risk of impaired function in fibrodysplasia ossificans progressiva (FOP)","volume":"109","author":"Pignolo","year":"2018","journal-title":"Bone."},{"key":"B4","doi-asserted-by":"publisher","first-page":"243","DOI":"10.1097\/00003086-199404000-00038","article-title":"Age- and joint-specific risk of initial heterotopic ossification in patients who have fibrodysplasia ossificans progressiva","volume":"301","author":"Rocke","year":"1994","journal-title":"Clin Orthop Relat Res."},{"key":"B5","doi-asserted-by":"publisher","first-page":"525","DOI":"10.1038\/ng1783","article-title":"A recurrent mutation in the BMP type I receptor ACVR1 causes inherited and sporadic fibrodysplasia ossificans progressiva","volume":"38","author":"Shore","year":"2006","journal-title":"Nat Genet."},{"key":"B6","doi-asserted-by":"publisher","first-page":"15438","DOI":"10.1073\/pnas.1510540112","article-title":"Neofunction of ACVR1 in fibrodysplasia ossificans progressiva","volume":"112","author":"Hino","year":"2015","journal-title":"Proc Natl Acad Sci USA."},{"key":"B7","doi-asserted-by":"publisher","first-page":"303ra137","DOI":"10.1126\/scitranslmed.aac4358","article-title":"ACVR1R206H receptor mutation causes fibrodysplasia ossificans progressiva by imparting responsiveness to activin A","volume":"7","author":"Hatsell","year":"2015","journal-title":"Sci Transl Med."},{"key":"B8","doi-asserted-by":"publisher","first-page":"629","DOI":"10.3390\/biomedicines9060629","article-title":"Activin-A induces early differential gene expression exclusively in periodontal ligament fibroblasts from fibrodysplasia ossificans progressiva patients","volume":"9","author":"Schoenmaker","year":"2021","journal-title":"Biomedicines."},{"key":"B9","doi-asserted-by":"publisher","first-page":"80","DOI":"10.1186\/1750-1172-6-80","article-title":"Fibrodysplasia ossificans progressiva: clinical and genetic aspects","volume":"6","author":"Pignolo","year":"2011","journal-title":"Orphanet J Rare Dis."},{"key":"B10","doi-asserted-by":"publisher","first-page":"650","DOI":"10.1002\/jbmr.2728","article-title":"The natural history of flare-ups in fibrodysplasia ossificans progressiva (FOP): a comprehensive global assessment","volume":"31","author":"Pignolo","year":"2016","journal-title":"J Bone Miner Res."},{"key":"B11","doi-asserted-by":"publisher","first-page":"909","DOI":"10.2106\/00004623-197961060-00019","article-title":"Fibrodysplasia ossificans progressiva. A survey of forty-two cases","volume":"61","author":"Rogers","year":"1979","journal-title":"J Bone Joint Surg Am."},{"key":"B12","doi-asserted-by":"publisher","first-page":"55","DOI":"10.1002\/jbm4.10008","article-title":"Flare-up after maxillofacial surgery in a patient with fibrodysplasia ossificans progressiva: an [F]-NaF PET\/CT study and a systematic review","volume":"2","author":"Eekhoff","year":"2018","journal-title":"JBMR Plus."},{"key":"B13","doi-asserted-by":"publisher","first-page":"76","DOI":"10.1302\/0301-620X.64B1.7068725","article-title":"Fibrodysplasia ossificans progressiva. The clinical features and natural history of 34 patients","volume":"64","author":"Connor","year":"1982","journal-title":"J Bone Joint Surg Br."},{"key":"B14","doi-asserted-by":"publisher","first-page":"239","DOI":"10.1385\/BMM:3:3-4:239","article-title":"Oral and dental health care and anesthesia for persons with fibrodysplasia ossificans progressiva","volume":"3","author":"Nussbaum","year":"2005","journal-title":"Clin Rev Bone Min Metab."},{"key":"B15","doi-asserted-by":"publisher","first-page":"21","DOI":"10.1016\/S1079-2104(96)80141-7","article-title":"Severe restriction in jaw movement after routine injection of local anesthetic in patients who have fibrodysplasia ossificans progressiva","volume":"81","author":"Luchetti","year":"1996","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod."},{"key":"B16","doi-asserted-by":"publisher","first-page":"204","DOI":"10.1111\/j.1601-0825.2005.01171.x","article-title":"Fibrodysplasia ossificans progressiva. An unusual cause of restricted mandibular movement","volume":"12","author":"van der Meij","year":"2006","journal-title":"Oral Dis."},{"key":"B17","doi-asserted-by":"publisher","first-page":"680","DOI":"10.1016\/j.tripleo.2003.08.002","article-title":"Ankylosis of the jaw in a patient with fibrodysplasia ossificans progressiva","volume":"96","author":"Herford","year":"2003","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod."},{"key":"B18","doi-asserted-by":"publisher","first-page":"143","DOI":"10.1186\/s12874-018-0611-x","article-title":"Systematic review or scoping review? Guidance for authors when choosing between a systematic or scoping review approach","volume":"18","author":"Munn","year":"2018","journal-title":"BMC Med Res Methodol."},{"key":"B19","doi-asserted-by":"publisher","first-page":"141","DOI":"10.1097\/XEB.0000000000000050","article-title":"Guidance for conducting systematic scoping reviews","volume":"13","author":"Peters","year":"2015","journal-title":"Int J Evid Based Healthc."},{"key":"B20","doi-asserted-by":"publisher","first-page":"75","DOI":"10.1213\/XAA.0000000000001152","article-title":"Successful airway and anesthesia management using a high-flow nasal cannula in a fibrodysplasia ossificans progressiva patient during general anesthesia: a case report","volume":"14","author":"Deguchi","year":"2020","journal-title":"A A Pract"},{"key":"B21","doi-asserted-by":"publisher","first-page":"543","DOI":"10.1111\/scd.12413","article-title":"Progressive ossificans fibrodysplasia endodontic management: case report","volume":"39","author":"Dutra","year":"2019","journal-title":"Spec Care Dentist."},{"key":"B22","doi-asserted-by":"publisher","first-page":"341","DOI":"10.1080\/08869634.2017.1349025","article-title":"Unresolving trismus following third molar surgery: report of a case of fibrodysplasia ossificans progressiva with review of literature","volume":"36","author":"Rajanikanth","year":"2018","journal-title":"Cranio."},{"key":"B23","doi-asserted-by":"publisher","first-page":"427","DOI":"10.1016\/j.bjoms.2018.03.021","article-title":"Severe trismus and contraindicated exodontia in a patient with fibrodysplasia ossificans progressiva: case report","volume":"56","author":"Geddis-Regan","year":"2018","journal-title":"Br J Oral Maxillofac Surg."},{"key":"B24","doi-asserted-by":"publisher","first-page":"184","DOI":"10.1016\/j.omsc.2018.09.005","article-title":"Fibrodysplasia ossificans progressive; its oral manifestations and management. A case-based discussion","volume":"4","author":"El Azem","year":"2018","journal-title":"Oral and Maxillofacial Surgery Cases."},{"key":"B25","doi-asserted-by":"publisher","first-page":"1891","DOI":"10.1016\/j.joms.2017.03.005","article-title":"Restricted mandibular movement attributed to ossification of mandibular depressors and medial pterygoid muscles in patients with fibrodysplasia ossificans progressiva: a report of 3 cases","volume":"75","author":"Okuno","year":"2017","journal-title":"J Oral Maxillofac Surg."},{"key":"B26","doi-asserted-by":"publisher","first-page":"31","DOI":"10.1016\/j.omsc.2016.09.001","article-title":"Dental extraction protocol in a patient with fibrodysplasia ossificans progressiva","volume":"2","author":"Haupt","year":"2016","journal-title":"Oral Maxillofac Surg Cases."},{"key":"B27","doi-asserted-by":"publisher","first-page":"37","DOI":"10.5812\/iranjradiol.10302","article-title":"Stone man: a case report","volume":"10","author":"Mortazavi","year":"2012","journal-title":"Iran J Radiol."},{"key":"B28","doi-asserted-by":"publisher","first-page":"404","DOI":"10.1016\/j.tripleo.2011.03.011","article-title":"Unusually severe limitation of the jaw attributable to fibrodysplasia ossificans progressiva: a case report with cone-beam computed tomography findings","volume":"113","author":"Orhan","year":"2012","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol."},{"key":"B29","doi-asserted-by":"publisher","first-page":"165","DOI":"10.1111\/j.1754-4505.2012.00260.x","article-title":"Facial morphology and occlusion of a patient with fibrodysplasia ossificans progressiva (FOP): a case report","volume":"32","author":"Susami","year":"2012","journal-title":"Spec Care Dentist."},{"key":"B30","doi-asserted-by":"publisher","first-page":"220","DOI":"10.1111\/j.1754-4505.2011.00216.x","article-title":"Improvement of mouth opening for a patient with fibrodysplasia ossificans progressiva: a case report","volume":"31","author":"Braga","year":"2011","journal-title":"Spec Care Dentist."},{"key":"B31","doi-asserted-by":"publisher","first-page":"11","DOI":"10.1016\/j.tripleo.2011.03.015","article-title":"Fibrodysplasia ossificans progressiva (FOP) in South Africa: dental implications in 5 cases","volume":"112","author":"Roberts","year":"2011","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod."},{"key":"B32","doi-asserted-by":"publisher","first-page":"1905","DOI":"10.1016\/j.joms.2010.12.011","article-title":"Extraction of 6 molars under general anesthesia in patient with fibrodysplasia ossificans progressiva","volume":"69","author":"Mori","year":"2011","journal-title":"J Oral Maxillofac Surg."},{"key":"B33","doi-asserted-by":"publisher","first-page":"69","DOI":"10.1097\/MCD.0b013e32833734f9","article-title":"Mandibular hypoplasia in fibrodysplasia ossificans progressiva causing obstructive sleep apnoea with pulmonary hypertension","volume":"19","author":"Carvalho","year":"2010","journal-title":"Clin Dysmorphol."},{"key":"B34","doi-asserted-by":"publisher","first-page":"499","DOI":"10.1016\/j.tripleo.2010.12.002","article-title":"Craniofacial findings in fibrodysplasia ossificans progressiva: computerized tomography evaluation","volume":"111","author":"Carvalho","year":"2011","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod."},{"key":"B35","doi-asserted-by":"publisher","first-page":"1408","DOI":"10.1016\/j.joms.2009.11.005","article-title":"Intraoral approach technique for treating trismus caused by fibrodysplasia ossificans progressiva","volume":"68","author":"Duan","year":"2010","journal-title":"J Oral Maxillofac Surg."},{"key":"B36","doi-asserted-by":"publisher","first-page":"106","DOI":"10.1111\/j.1754-4505.2010.00133.x","article-title":"A description of two surgical and anesthetic management techniques used for a patient with fibrodysplasia ossificans progressiva","volume":"30","author":"Wadenya","year":"2010","journal-title":"Spec Care Dentist."},{"key":"B37","doi-asserted-by":"publisher","first-page":"684","DOI":"10.1111\/j.1460-9592.2005.01837.x","article-title":"Anesthesia in a child with fibrodysplasia ossificans progressiva","volume":"16","author":"Vashisht","year":"2006","journal-title":"Paediatr Anaesth."},{"key":"B38","doi-asserted-by":"publisher","first-page":"312","DOI":"10.1016\/j.tripleo.2005.09.020","article-title":"Severe limitation in jaw movement in a patient with fibrodysplasia ossificans progressiva: a case report","volume":"102","author":"Sendur","year":"2006","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod."},{"key":"B39","doi-asserted-by":"publisher","first-page":"1238","DOI":"10.1097\/00006534-199904010-00020","article-title":"Fibrodysplasia ossificans progressiva and associated osteochondroma of the coronoid process in a child","volume":"103","author":"Chichareon","year":"1999","journal-title":"Plast Reconstr Surg."},{"key":"B40","doi-asserted-by":"publisher","first-page":"204","DOI":"10.1016\/S0278-2391(10)80212-5","article-title":"Failure of surgery and isotretinoin to relieve jaw immobilization in fibrodysplasia ossificans progressiva: report of two cases","volume":"48","author":"Crofford","year":"1990","journal-title":"J Oral Maxillofac Surg."},{"key":"B41","doi-asserted-by":"publisher","first-page":"117","DOI":"10.1016\/0007-117X(82)90018-X","article-title":"Extra-articular ankylosis in fibrodysplasia ossificans progressiva","volume":"20","author":"Connor","year":"1982","journal-title":"Br J Oral Surg."},{"key":"B42","doi-asserted-by":"publisher","first-page":"31","DOI":"10.1016\/0007-117X(82)90004-X","article-title":"Abnormal temporomandibular joints in fibrodysplasia ossificans progressiva","volume":"20","author":"Renton","year":"1982","journal-title":"Br J Oral Surg."},{"key":"B43","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1111\/j.1754-4505.2007.tb01748.x","article-title":"Oral management in a patient with fibrodysplasia ossificans progressiva","volume":"27","author":"Young","year":"2007","journal-title":"Spec Care Dentist."},{"key":"B44","doi-asserted-by":"publisher","first-page":"668","DOI":"10.1016\/j.joms.2008.07.011","article-title":"Submandibular space swelling in a patient with fibrodysplasia ossificans progressiva: a diagnostic dilemma","volume":"67","author":"Leavitt","year":"2009","journal-title":"J Oral Maxillofac Surg."},{"key":"B45","doi-asserted-by":"publisher","first-page":"856","DOI":"10.1016\/j.jcms.2013.01.044","article-title":"Fibrodysplasia ossificans progressiva (FOP): report of a case with extra-articular ankylosis of the mandible","volume":"41","author":"Kriegbaum","year":"2013","journal-title":"J Craniomaxillofac Surg."},{"key":"B46","doi-asserted-by":"publisher","first-page":"932","DOI":"10.1002\/ca.20865","article-title":"An anatomical study of the muscles that attach to the articular disc of the temporomandibular joint","volume":"22","author":"Matsunaga","year":"2009","journal-title":"Clin Anat."},{"key":"B47","doi-asserted-by":"publisher","first-page":"1072","DOI":"10.1053\/joms.2002.34424","article-title":"Myositis ossificans traumatica of masticatory musculature: A case report and literature review","volume":"60","author":"Kim","year":"2002","journal-title":"J Oral Maxillofac Surg."}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2022.852678\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,3,27]],"date-time":"2022-03-27T21:05:59Z","timestamp":1648415159000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2022.852678\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,3,22]]},"references-count":47,"alternative-id":["10.3389\/fmed.2022.852678"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2022.852678","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,3,22]]},"article-number":"852678"}}