Endodontic Management Of A Patient With X‐linked Hypophosphataemic Rickets

Alexander, Stanley A.; Moloney, Luke; Kilpatrick, Nicky

doi:10.1111/j.1747-4477.2001.tb00340.x

Cited by 6 publications

(16 citation statements)

References 11 publications

(3 reference statements)

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“…It is an unusual presentation to find pulpal and apical pathosis in intact teeth. Yet, this presentation is consistent with other published case reports of patients with XLH (Murayama et al 2000, Alexander et al 2001, Pereira et al 2004, Beltes & Zachou 2012, Lee et al 2017, James & Roudsari 2019.…”

Section: Discussionsupporting

confidence: 92%

“…Patients with XLH may present with dental abnormalities and complications both in the primary and permanent dentition. Published case reports and case series have consistently demonstrated spontaneous pulpal necrosis occurring in multiple teeth in the absence of caries or trauma (Goodman et al 1998, Murayama et al 2000, Alexander et al 2001, Pereira et al 2004, Batra et al 2006, Douyere et al 2009, Beltes & Zachou 2012, Soares et al 2013, Lee et al 2017, James & Roudsari 2019. Andersen et al (2012) found patients with XLH had a characteristically large number of endodontically affected teeth with a mean of 4.2.…”

Section: Introductionmentioning

confidence: 99%

“…Clinically, enamel defects have been reported, including hypoplasia and infractions (Goodman et al 1998, Souza et al 2010, Rabbani et al 2012. Radiographically, a range of abnormal morphological features may be present, including enlarged pulp chambers and pulp horns, enlarged root canals, short roots and open apices (Hillmann & Geurtsen 1996, Goodman et al 1998, Murayama et al 2000, Alexander et al 2001, Pereira et al 2004, Souza et al 2010, Lee et al 2017. Histologically, enamel has occasionally been reported as thin and microcracks extending to the amelo-dentinal junction have also been identified (Douyere et al 2009, Opsahl Vital et al 2012.…”

Section: Introductionmentioning

confidence: 99%

“…Root canal treatment (RCT) appears to be the treatment of choice for the majority of permanent teeth with spontaneous pulpal necrosis (Murayama et al 2000, Alexander et al 2001, Pereira et al 2004, Beltes & Zachou 2012, Lee et al 2017. Despite the abnormal morphology, the majority of case reports used either a cold lateral condensation or a continuous wave technique; however, radiographic follow-up was not always presented (Alexander et al 2001, Beltes & Zachou 2012, Lee et al 2017.…”

Section: Introductionmentioning

confidence: 99%

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Presentation and non‐surgical endodontic treatment of two patients with X‐linked hypophosphatemia: a case report

Bradley¹,

Dutta

Philpott³

2021

Int Endodontic J

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Aim 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. Summary 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 followup, 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.

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Section: Discussionsupporting

confidence: 92%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Presentation and non‐surgical endodontic treatment of two patients with X‐linked hypophosphatemia: a case report

Bradley¹,

Dutta

Philpott³

2021

Int Endodontic J

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“…The literature on dental manifestations in HR patients consists of more than 60 case reports or case series with small numbers of cases of which, however, only a few have been published in 2000 or later (3–16). Thus, many studies were performed before the genetic analysis was available.…”

Section: Introductionmentioning

confidence: 99%

Periapical and endodontic status of permanent teeth in patients with hypophosphatemic rickets

Andersen

Beck-Nielsen

Haubek

et al. 2011

J of Oral Rehabilitation

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Hypophosphatemic rickets (HR) is a rare hereditary disease in which dental problems in terms of spontaneous periapical infections are frequently reported. Most previous reports have been based on a small number of HR patients and have been published before the disease could be confirmed genetically. The aim of the present study was to describe the periapical and endodontic status of permanent teeth in patients with genetically and/or biochemically confirmed HR. The patients were recruited from a medical study on HR patients. The patients underwent a dental examination including a digital panoramic radiograph, which was scored for endodontically affected teeth (i.e. teeth with periapical radiolucencies and/or endodontically treated teeth). A total of 52 patients (age range: 5·7-74·5 years; 17 males and 35 females) were included. HR patients were characterised by a high number of endodontically affected teeth (mean: 4·2; s.d.: 5·0). The number of affected teeth rose significantly with age (P < 0·01), and no statistically significant gender difference was found. The relative distribution of endodontically affected teeth in the three tooth groups (incisors and canines, premolars, and molars) varied according to age. In the youngest age group, only incisors and canines were affected, while the relative proportion of affected premolars and molars increased with age. Endodontically affected teeth are common in HR patients, and the number of affected teeth increased significantly with age. Hence, the need for endodontic treatment among HR patients is comprehensive.

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