Generalized radiculomegaly and macrodontic dental appearance in a patient with common variable immunodeficiency: a case report.
4 affiliations
- 1Department of Oral and Maxillofacial Surgery, Institute of Dental Sciences, Siksha 'O' Anusandhan University, Bhubaneswar, India. [email protected].
- 2Department of Oral and Maxillofacial Surgery, Institute of Dental Sciences, Siksha 'O' Anusandhan University, Bhubaneswar, India.
- 3Department of Hematology, Institute of Medical Sciences and SUM Hospital, Siksha 'O' Anusandhan University, Bhubaneswar, India.
- 4Department of Rheumatology and Immunology, Institute of Medical Sciences and SUM Hospital, Siksha 'O' Anusandhan University, Bhubaneswar, India.
Abstract
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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BibTeX
@article{medhi2026generalized,
title = {{Generalized radiculomegaly and macrodontic dental appearance in a patient with common variable immunodeficiency: a case report.}},
author = {Upasa Sarma Medhi and Abhinn Miglani and Subrat Kumar Padhiary and Priyanka Samal and Pradeepta Sekhar Patro},
journal = {Journal of medical case reports},
year = {2026},
month = aug,
doi = {10.1186/s13256-026-06529-3},
url = {https://europepmc.org/article/MED/42958379},
note = {CC BY-NC-ND},
}
RIS
TY - JOUR
TI - Generalized radiculomegaly and macrodontic dental appearance in a patient with common variable immunodeficiency: a case report.
AU - Upasa Sarma Medhi
AU - Abhinn Miglani
AU - Subrat Kumar Padhiary
AU - Priyanka Samal
AU - Pradeepta Sekhar Patro
PY - 2026
DA - 2026/08/20
JO - Journal of medical case reports
DO - 10.1186/s13256-026-06529-3
UR - https://europepmc.org/article/MED/42958379
AB - 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).
SN - 1752-1947
ER -