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Cherubism--a case report with long term follow up
Lakshmi V Chavali1, R M Rao Bhimalingam, P V Sudhakar
1Department of Pathology, Rangaraya Medical College, Kakinada, Andhra Pradesh, India. drcvlakshmi@gmail.com
Indian Journal of Pathology & Microbiology
|January 12, 2012
Summary
Cherubism, a rare jaw disorder, presents with painless swelling. This case study highlights its natural progression, from initial mandibular lesions to maxillary involvement, eventually regressing post-puberty.
Area of Science:
- Oral and Maxillofacial Surgery
- Genetics
- Pathology
Background:
- Cherubism is a rare, non-neoplastic bone disease causing bilateral jaw enlargement.
- It typically presents with a characteristic cherubic facial appearance.
- While often autosomal dominant, sporadic cases also occur.
Observation:
- A 7-year-old male presented with bilateral mandibular swellings.
- Radiological imaging revealed multilocular lytic lesions in the mandible.
- Histopathology showed proliferating fibrous connective tissue with multinucleated giant cells.
Findings:
- The nonfamilial cherubism case demonstrated disease progression, including maxilla involvement after 5 years.
- Surgical intervention was performed for the maxilla.
- Remarkably, lesions showed spontaneous regression with improved facial contour by 11-year follow-up.
Implications:
- This case illustrates the natural history of cherubism, including progression, stabilization, and post-pubertal involution.
- Highlights the importance of long-term follow-up for managing cherubism.
- Further research into cherubism pathogenesis is warranted to improve understanding and treatment.
