Radiculomegaly: a case report of this rare dental finding with review of the associated oculo-facio-cardio-dental syndrome

Smith, Molly Housley; Cohen, Donald M.; Bhattacharyya, Indraneel; Islam, Nadim; Kashtwari, D.

doi:10.1016/j.oooo.2018.02.011

Cited by 8 publications

(12 citation statements)

References 41 publications

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“…In the present case, an association between radiculomegaly and OFCD was excluded since OFCD is an Xchromosome linked disease and is fatal in males [4,10,12]. Our patient also did not exhibit any eye or heart problems.…”

Section: Discussionmentioning

confidence: 55%

“…However, radiculomegaly was later reported as a characteristic feature of males affected by Klinefelter syndrome [3,8]. To date, only a few nonsyndromic cases of radiculomegaly have been reported [9,10]. Here, we report a case of nonsyndromic radiculomegaly in a Saudi individual and then discuss the aspects of this case in relation to previous studies (Table 1).…”

Section: Introductionmentioning

confidence: 60%

“…In the present case, an extreme variation in root length was detected in periapical radiographs (PAs) and with CBCT. Among published reports of radiculomegaly, canine teeth are most commonly affected [4,9,10,20,21]. Being able to obtain accurate root canal length measurements after treatment is also essential for determining the success of a treatment.…”

Section: Discussionmentioning

confidence: 99%

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A Rare Case of Nonsyndromic Generalized Radiculomegaly with a Literature Review

Al-Obaida

2020

Case Reports in Dentistry

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Radiculomegaly is a rare condition involving elongated tooth roots. This condition has significant clinical implications and has been associated with syndromes such as oculofaciocardiodental syndrome. However, only a few nonsyndromic cases of radiculomegaly have been reported. Here, we report a case of nonsyndromic radiculomegaly in a Saudi individual. A subsequent review of the literature suggests endodontic management modalities for individuals with the dental findings demonstrated in the present case.

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Section: Discussionmentioning

confidence: 55%

Section: Introductionmentioning

confidence: 60%

Section: Discussionmentioning

confidence: 99%

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A Rare Case of Nonsyndromic Generalized Radiculomegaly with a Literature Review

Al-Obaida

2020

Case Reports in Dentistry

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“…The first reports of radiculomegaly [ 5 – 7 ] related it as a characteristic feature of the X-linked rare congenital syndrome, oculofaciocardiodental syndrome (OFCD). OFCD syndrome is an X-linked dominant trait with potential male lethality [ 4 , 8 , 9 ], which affects the ocular, facial, cardiac, and dental aspects, with an incidence of less than 1 in 1 million people [ 4 , 8 – 11 ]. Among different dental anomalies that can be found with OFCD syndrome, radiculomegaly of the canines is highly related to this syndrome [ 4 , 8 , 12 – 14 ].…”

Section: Introductionmentioning

confidence: 99%

Nonsyndromic Generalized Radiculomegaly of Permanent Dentition: A Rare Case Report

Alhussain

Almosa

Alnofaie

2022

Case Reports in Dentistry

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Introduction. Radiculomegaly (marked elongation of dental roots) is a distinct dental abnormality with a major clinical significance that is closely related to oculofaciocardiodental syndrome (OFCD). Since OFCD syndrome was first identified in 1996, only a few cases of nonsyndromic/nonfamilial radiculomegaly have been reported. We report a new nonsyndromic/nonfamilial radiculomegaly case and the association of OFCD syndrome with the dental challenges. Case Presentation. 18-year-old medically free male presented to the screening dental clinics complaining of teeth malposition. Panoramic radiograph incidentally revealed extremely long and wide roots of almost all dentition. Apical radiographs and cone-beam computed tomography (CBCT) were taken to confirm this anomaly and to rule out any artifact. The images confirmed the excessive length of the roots. A cephalometric X-ray was performed on the patient to rule out any dentofacial deformity. History and physical examinations were negative for ocular or cardiac anomalies. Due to lack of evidence of physical signs and symptoms consistent with OFCD syndrome, genetic testing was not performed. Conclusion. Dentists need to be informed of the clinical and radiographic features of different dental anomalies, especially radiculomegaly, as it is considered one of the significant features of OFCD syndrome. Challenges related to radiculomegaly face dentists while the current literature has not yet provided a solid baseline for managing such patients. The challenges include repairing the root canals, extracting and/or moving the teeth orthodontically. Early diagnosis of the syndrome is crucial to prevent dental challenges and provide the best dental care services.

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“…According to the authors, the typical facial features include a long narrow face, a high nasal bridge, and a bifid nasal tip. The dental anomalies include radiculomegaly, oligodontia, root dilacerations, malocclusion, and delayed eruptions (Gorlin et al, 1996;Kato, Kushima e Kushima, 2018;). Smith, Cohen, Bhattacharyya, Islam, & Kashtwari (2018 reviewed the literature concerning the patients with radiculomegaly and OFCD syndrome, revealing that in only one study was confirmed a case of nonsyndromic/nonfamilial radiculomegaly until that moment.…”

Section: Introductionmentioning

confidence: 99%

Technical adaptations of manual and rotary instruments for preparing a giant canine tooth

Stroparo

Moura³

et al. 2021

RSD

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Teeth with extraordinary length are a rare condition that complicates endodontic procedures due to the difficult in being reached the working length and making the adequate preparation of the root canal, once there are no endodontic instruments longer than 31 mm commercially available. This study presents a description of technical adaptations for endodontic preparation of a maxillary canine with 39.59 mm of length. The procedures adopted were based on the consultation of the literature regarding the treatment of long teeth, and included endodontic instrument adaptation and changing the reference point of the instrument. This study shows that extremely long teeth can be successfully instrumented endodontically using the described clinical techniques.

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Radiculomegaly: a case report of this rare dental finding with review of the associated oculo-facio-cardio-dental syndrome

Cited by 8 publications

References 41 publications

A Rare Case of Nonsyndromic Generalized Radiculomegaly with a Literature Review

A Rare Case of Nonsyndromic Generalized Radiculomegaly with a Literature Review

Nonsyndromic Generalized Radiculomegaly of Permanent Dentition: A Rare Case Report

Technical adaptations of manual and rotary instruments for preparing a giant canine tooth

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