Department of Restorative Dentistry, Edinburgh Dental Institute, Edinburgh, UK.
Department of Restorative Dentistry, School of Dentistry, College of Biomedical and Life Sciences, Cardiff University, Cardiff, UK.
Int Endod J. 2021 Aug;54(8):1403-1414. doi: 10.1111/iej.13520. Epub 2021 May 1.
To describe two patients with X-linked 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.
Two young male patients with X-linked 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- and 5-year 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-year period. Patient 2 underwent root canal treatment on 10 permanent teeth over a 5-year period. At follow-up, 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-up of 4.5 years.
Patients with X-linked hypophosphatemia may present with 'spontaneous' signs of pulp necrosis in multiple teeth in the absence of caries and trauma posing a diagnostic challenge. Abnormal morphological features, including wide canals and open apices, may present challenges during root canal treatment. Outcomes demonstrate that an appropriate root canal treatment protocol, including the application of apexification procedures, may be implemented to successfully manage such cases.
描述两例 X 连锁低磷血症患者，他们在多颗完整的牙齿中出现自发性牙髓坏死迹象。本文讨论了其临床表现和治疗方法，以及诊断和根管治疗的挑战。
两名患有 X 连锁低磷血症的年轻男性患者因牙齿感染问题被转诊至英国爱丁堡牙科学院修复牙科。这两名患者均因不典型的临床表现和异常的根管形态而被转诊。在 3 至 5 年的治疗过程中，他们多次因疼痛或窦道而就诊。临床检查发现，牙齿完整，颊侧肿胀，颊侧窦道引流，牙髓活力测试呈阴性。影像学检查通常包括根尖片，结果显示牙齿完整，存在多种异常形态特征，包括较大的牙髓腔、宽阔的根管、短根和开放的根尖，所有这些都伴有根尖周透光区。由于形态异常，一些牙齿需要使用三氧化矿物聚合体（mineral trioxide aggregate，MTA）进行根尖诱导成形术。患者 1 的 21 号和 43 号牙在 3 年内接受了根管治疗。患者 2 的 10 颗恒牙在 5 年内接受了根管治疗。随访时，两名患者均无症状，临床检查牙齿无感染或根尖炎症迹象。影像学检查证实 12 颗牙中有 11 颗的根尖透光区完全消失。随访时间长达 4.5 年，结果均显示良好。
X 连锁低磷血症患者可能在无龋病和外伤的情况下出现多颗牙齿的“自发性”牙髓坏死迹象，这对诊断构成挑战。异常的形态特征，包括宽阔的根管和开放的根尖，可能在根管治疗过程中带来挑战。研究结果表明，实施适当的根管治疗方案，包括根尖诱导成形术，可能有助于成功治疗此类病例。
