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

Publication date: Available online 28 February 2018
Source:Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Author(s): Molly Housley Smith, Donald M. Cohen, Indraneel Bhattacharyya, Nadim M. Islam, Deeba Kashtwari
Background: Radiculomegaly, or root gigantism, is a rare dental abnormality with important clinical implications. It is highly specific for oculo-facio-cardio-dental syndrome (OFCD), which places dentists at the forefront of diagnosis for this syndrome. Only one case of non-syndromic radiculomegaly has been reported in the literature since the description of OFCD in 1996. We present the second confirmed, non-syndromic/non-familial case and review the literature for dental treatments in patients with this dental finding. Methods: A review of the English-language literature was performed within PubMed for patients with radiculomegaly or OFCD. Teeth affected by radiculomegaly, gender, oro-dental findings, presence of OFCD syndrome, and dental treatment methods were recorded. Results: Sixty-seven cases of radiculomegaly and 92 OFCD cases were found in the literature. Only one confirmed case of non-syndromic/non-familial radiculomegaly existed prior to our report. Ten cases reported dental treatment or treatment plan details, and even fewer detailed the specific methods. Conclusions: Because dental anomalies, especially radiculomegaly, are a primary feature of OFCD, dentists should be aware of the clinical and radiographic features. Radiculomegaly poses a distinct challenge to dentists, and reports of dental therapy provided to these patients are sparse. Early diagnosis of the syndrome may prevent dental challenges and improve prognosis.



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