Generalized radiculomegaly and macrodontic dental appearance in a patient with common variable immunodeficiency: a case report
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Published on: August 22, 2022
Upasa Sarma Medhi1, Abhinn Miglani2, Subrat Kumar Padhiary2
1Department of Oral and Maxillofacial Surgery, Institute of Dental Sciences, Siksha 'O' Anusandhan University, Bhubaneswar, India. upasa1234@gmail.com.
Background:
Common variable immunodeficiency (CVID) is a primary immunodeficiency disorder characterized by hypogammaglobulinemia and impaired antibody production, predisposing affected individuals to recurrent infections and immune dysregulation. Although oral infections and periodontal disease have been reported in patients with CVID, developmental dentofacial abnormalities have rarely been described.
Case Presentation:
A 19-year-old male with a previously established diagnosis of CVID presented with pain, swelling, and purulent discharge from the chin region secondary to a chronic odontogenic sinus tract. Clinical examination revealed generalized enlargement of the dentition with marked crowding, producing a macrodontic dental appearance. Radiographic and cone-beam computed tomography (CBCT) evaluation demonstrated generalized root elongation involving both anterior and posterior teeth. The diagnosis of generalized radiculomegaly was supported by radiographic evidence of generalized root elongation and measurements exceeding available published reference values for the tooth types for which appropriate normative data were available. The patient also exhibited microcytic hypochromic anemia, mild coagulation abnormalities, recurrent sino-respiratory infections, bronchiectasis, conductive hearing loss, and elevated alkaline phosphatase levels. Surgical management of recurrent odontogenic infection was successfully achieved through multidisciplinary coordination involving oral and maxillofacial surgeons, hematologists, and immunologists.
Conclusion:
To the best of our knowledge, generalized radiculomegaly has not previously been reported in association with CVID. Although the coexistence of CVID and generalized root elongation is noteworthy, a direct causal association cannot be established from a single case. Further clinical and genetic studies are required to determine whether this represents a true association. Additional clinical and genetic studies are required to clarify the significance of this unusual dentofacial phenotype. This case report was prepared in accordance with the CARE guidelines 2013 (Gagnier, J. J., et al., 2013).
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