{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,2]],"date-time":"2026-07-02T22:58:58Z","timestamp":1783033138546,"version":"3.54.6"},"reference-count":34,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2021,5,1]],"date-time":"2021-05-01T00:00:00Z","timestamp":1619827200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Int Endodontic J"],"published-print":{"date-parts":[[2021,8]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Aim<\/jats:title><jats:p>To describe two patients with X\u2010linked hypophosphatemia presenting with spontaneous signs of pulpal necrosis in multiple intact teeth. The presentation and management are discussed, along with the diagnostic and endodontic treatment challenges.<\/jats:p><\/jats:sec><jats:sec><jats:title>Summary<\/jats:title><jats:p>Two young male patients with X\u2010linked hypophosphatemia were referred to the Department of Restorative Dentistry at the Edinburgh Dental Institute, UK for management of dental infection. Both patients were referred due to their unusual clinical presentation and abnormal root canal morphology. They subsequently presented on multiple occasions with pain or sinus tracts over a 3\u2010 and 5\u2010year period whilst under care. Clinical examination revealed intact teeth with buccal swellings, draining buccal sinuses and negative responses to pulp sensibility testing. Radiographic examination, generally consisting of periapical radiographs, revealed intact teeth with a range of unusual morphological features including large pulp chambers, wide canals, short roots and open apices, all with associated periapical radiolucencies. Due to the unusual morphology, some teeth required apexification with a mineral trioxide aggregate plug. Patient 1 underwent root canal treatment on teeth 21 and 43 over a 3\u2010year period. Patient 2 underwent root canal treatment on 10 permanent teeth over a 5\u2010year period. At follow\u2010up, both patients were asymptomatic and clinically the teeth had no signs of infection or periapical inflammation. Radiographic examination confirmed complete resolution of the apical radiolucencies on 11 out of 12 teeth. Favourable outcomes have been demonstrated up to a follow\u2010up of 4.5\u00a0years.<\/jats:p><\/jats:sec><jats:sec><jats:title>Key learning points<\/jats:title><jats:p>\n<jats:list list-type=\"bullet\">\n\n<jats:list-item><jats:p>Patients with X\u2010linked hypophosphatemia may present with \u2018spontaneous\u2019 signs of pulp necrosis in multiple teeth in the absence of caries and trauma posing a diagnostic challenge.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Abnormal morphological features, including wide canals and open apices, may present challenges during root canal treatment.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Outcomes demonstrate that an appropriate root canal treatment protocol, including the application of apexification procedures, may be implemented to successfully manage such cases.<\/jats:p><\/jats:list-item>\n<\/jats:list>\n<\/jats:p><\/jats:sec>","DOI":"10.1111\/iej.13520","type":"journal-article","created":{"date-parts":[[2021,3,22]],"date-time":"2021-03-22T13:16:53Z","timestamp":1616419013000},"page":"1403-1414","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Presentation and non\u2010surgical endodontic treatment of two patients with X\u2010linked hypophosphatemia: a case report"],"prefix":"10.1111","volume":"54","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3822-3934","authenticated-orcid":false,"given":"H.","family":"Bradley","sequence":"first","affiliation":[{"name":"Department of Restorative Dentistry Edinburgh Dental Institute Edinburgh UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Dutta","sequence":"additional","affiliation":[{"name":"Department of Restorative Dentistry School of Dentistry College of Biomedical and Life Sciences 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