{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,15]],"date-time":"2026-09-15T07:11:21Z","timestamp":1789456281116,"version":"build-2803163510"},"reference-count":13,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2017,7,5]],"date-time":"2017-07-05T00:00:00Z","timestamp":1499212800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JBMR Plus"],"published-print":{"date-parts":[[2018,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:sec><jats:label\/><jats:p>Fibrodysplasia ossificans progressiva (FOP) is a rare genetic disorder leading to progressive heterotopic ossifications (HO) of muscles, tendons, and ligaments, which can be induced by trauma or by surgery. Despite strong medical advice to the contrary, an FOP patient insisted on surgery to alleviate her complete trismus, which caused an unbearable impact on her quality of life (QOL). The entire trismus history of this FOP patient is presented. [<jats:sup>18<\/jats:sup>F]\u2010NaF position emission tomography\/computed tomography (PET\/CT) scans were introduced as an imaging method for heterotopic bone formation activity. To place our findings into context, a systematic review on jaw surgery in FOP was performed. After falling down the stairs, a 9\u2010year\u2010old patient developed mobility impairment of her left\u2010sided jaw. During the following 13 years bone scintigraphy showed persistent activity of the disease leading to progressive left\u2010sided zygomatico\u2010mandibular fusion by HO, resulting in complete trismus. Within 1 month after HO removal on the left side and a matching right coronoidectomy, [<jats:sup>18<\/jats:sup>F]\u2010NaF PET\/CT demonstrated a substantial flare\u2010up activity followed by new HO in both masseter and temporalis muscles. Despite recurrent HO and trismus her QOL increased due to a stable increased interincisal opening of 5.5\u2009mm. Although systematic review reveals a 100% risk of HO recurrence after jaw surgery, information on improved QOL is scarce. In conclusion, surgery in FOP may be beneficial for QOL despite new HO formation. Assessment of disease activity using [<jats:sup>18<\/jats:sup>F]\u2010NaF PET\/CT is possible before HO is evident on CT and may serve as a new and quantitative marker of the disease. \u00a9 2017 The Authors. <jats:italic>JBMR Plus<\/jats:italic> Published by Wiley Periodicals, Inc. on behalf of the American Society for Bone and Mineral Research.<\/jats:p><\/jats:sec>","DOI":"10.1002\/jbm4.10008","type":"journal-article","created":{"date-parts":[[2017,5,26]],"date-time":"2017-05-26T13:03:22Z","timestamp":1495803802000},"page":"55-58","source":"Crossref","is-referenced-by-count":26,"title":["Flare\u2010Up After Maxillofacial Surgery in a Patient With Fibrodysplasia Ossificans Progressiva: An [<sup>18<\/sup>F]\u2010NaF PET\/CT Study and a Systematic Review"],"prefix":"10.1093","volume":"2","author":[{"given":"E Marelise W","family":"Eekhoff","sequence":"first","affiliation":[{"name":"Department of Internal Medicine Section Endocrinology VU University Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J Coen","family":"Netelenbos","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine Section Endocrinology VU University Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pim","family":"de Graaf","sequence":"additional","affiliation":[{"name":"Department of Radiology &amp; Nuclear Medicine VU University Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Max","family":"Hoebink","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine Section Endocrinology VU University Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie","family":"Bravenboer","sequence":"additional","affiliation":[{"name":"Department of Clinical Genetics and Bone Histomorphology VU University Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dimitra","family":"Micha","sequence":"additional","affiliation":[{"name":"Department of Clinical Genetics and Bone Histomorphology VU University Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gerard","family":"Pals","sequence":"additional","affiliation":[{"name":"Department of Clinical Genetics and Bone Histomorphology VU University Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teun J","family":"de Vries","sequence":"additional","affiliation":[{"name":"Department Periodontology Academic Centre for Dentistry Amsterdam (ACTA) University of Amsterdam and VU University Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adriaan A","family":"Lammertsma","sequence":"additional","affiliation":[{"name":"Department of Radiology &amp; 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