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

Oral Surg Oral Med Oral Pathol Oral Radiol. 2018 Oct;126(4):e220-e227. doi: 10.1016/j.oooo.2018.02.011. Epub 2018 Feb 28.

Abstract

Background: Radiculomegaly, or root gigantism, is a rare dental abnormality with important clinical implications. It is highly specific for oculo-facio-cardio-dental (OFCD) syndrome, which places dentists at the forefront of diagnosis of this syndrome. Only 1 case of nonsyndromic radiculomegaly has been reported in the literature since the description of OFCD syndrome in 1996. We present the second confirmed, nonsyndromic/nonfamilial case and review the literature for dental treatments in patients with this dental finding.

Study design: A review of the English language literature was performed in PubMed for patients with radiculomegaly or OFCD syndrome. Teeth affected by radiculomegaly, gender, orodental findings, presence of OFCD syndrome, and dental treatment methods were recorded.

Results: Sixty-seven cases of radiculomegaly and 92 cases of OFCD syndrome were found in the literature. Only 1 confirmed case of nonsyndromic/nonfamilial radiculomegaly had been reported previously. Ten reports described dental treatment or treatment plan details, and even fewer included specific methods.

Conclusions: Because dental anomalies, especially radiculomegaly, are a primary feature of OFCD syndrome, 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.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Cataract / congenital*
  • Cataract / diagnosis
  • Diagnosis, Differential
  • Female
  • Heart Septal Defects / diagnosis*
  • Humans
  • Microphthalmos / diagnosis*
  • Tooth Root / abnormalities*
  • Tooth Root / diagnostic imaging
  • Young Adult

Supplementary concepts

  • Microphthalmia, syndromic 2