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Unicentric Castleman Disease Involving the Submandibular Gland: A Case Report
Xinlong Sun1, Pengfei Dong2, Beining Zhang1
1Department of Oral and Maxillofacial Surgery, Yantai Affiliated Hospital of Binzhou Medical University, Yantai, China, byytfy.com.
Case Reports in Dentistry
|August 14, 2026
Summary
Unicentric Castleman disease (UCD) is rare in the submandibular region. This case highlights UCD diagnosis via imaging and histology, with successful surgical cure and no recurrence.
Area of Science:
- Pathology
- Oncology
- Head and Neck Surgery
Background:
- Castleman disease is a rare lymphoproliferative disorder.
- Unicentric Castleman disease (UCD) typically presents as a localized lymph node enlargement.
- Submandibular gland involvement by UCD is exceptionally rare.
Purpose of the Study:
- To report a rare case of unicentric Castleman disease involving the submandibular gland and lymph nodes.
- To emphasize the diagnostic features and successful management of this rare presentation.
- To increase awareness of UCD in the differential diagnosis of submandibular masses.
Main Methods:
- Case presentation of a 56-year-old male with a submandibular mass.
- Diagnostic imaging revealing well-defined heterogeneous structures with prominent vascularity.
- Excisional biopsy with histopathological and immunohistochemical analysis.
Main Results:
- Histopathology confirmed the hyaline-vascular variant of Castleman disease.
- Features included atrophic germinal centers with "onion-skin" layering and prominent vascularity.
- Complete surgical excision resulted in a disease-free status at 3-year follow-up.
Conclusions:
- Unicentric Castleman disease can manifest in the submandibular region.
- Characteristic imaging and histopathology aid in diagnosis.
- Complete surgical resection is a curative treatment for localized UCD.

