Adult-Onset Xanthogranulomatous Inflammation Presenting as a Cervical Branchial-Cleft Cyst

Xanthogranulomatous inflammation (XGI) is a rare benign histiocytic reaction in the head and neck. Adult-onset XGI within a branchial-cleft cyst (BCC) is exceptionally uncommon, with only two cases previously reported. We present the oldest documented patient and review the literature. A 71-year-old man presented with a painless 3-cm right lateral-neck mass. Ultrasound and MRI suggested a benign thin-walled cyst; fine-needle aspiration revealed foamy macrophages without atypia. Intraoperatively, dense fibrosis and firm adhesion to the carotid sheath raised concern for metastatic carcinoma. Frozen-section analysis demonstrated foamy histiocytes and Touton giant cells without epithelial atypia, confirming XGI. En-bloc excision was completed without radical neck dissection. Recovery was uneventful, and no recurrence was observed at 14 months. Only three adult BCC-XGI cases have been reported, including the present patient. All showed deceptively benign imaging, disproportionate intraoperative fibrosis, and reliance on frozen section to exclude malignancy. Chronic leakage or rupture of cyst contents - possibly potentiated by fine-needle aspiration - appears to trigger the xanthogranulomatous response. Surgeons should consider XGI when encountering fibrotic lateral cervical cysts in older adults. Prompt intraoperative frozen-section analysis enables conservative excision and prevents overtreatment.

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Journal
Cureus
Published
2025-06-02
DOI
https://doi.org/10.7759/cureus.85215
Primary Topic
Infectious Disease Case Reports and Treatments
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article
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article

Adult-Onset Xanthogranulomatous Inflammation Presenting as a Cervical Branchial-Cleft Cyst

Shinichi Oikawa, Ryoukichi Ikeda, Yoshihiko Koike, Daiki Onodera et al.
Cureus
Infectious Disease Case Reports and Treatments
article

Adult-Onset Xanthogranulomatous Inflammation Presenting as a Cervical Branchial-Cleft Cyst

Shinichi Oikawa, Ryoukichi Ikeda, Yoshihiko Koike, Daiki Onodera, Aya Katsura
article en

Abstract

Xanthogranulomatous inflammation (XGI) is a rare benign histiocytic reaction in the head and neck. Adult-onset XGI within a branchial-cleft cyst (BCC) is exceptionally uncommon, with only two cases previously reported. We present the oldest documented patient and review the literature. A 71-year-old man presented with a painless 3-cm right lateral-neck mass. Ultrasound and MRI suggested a benign thin-walled cyst; fine-needle aspiration revealed foamy macrophages without atypia. Intraoperatively, dense fibrosis and firm adhesion to the carotid sheath raised concern for metastatic carcinoma. Frozen-section analysis demonstrated foamy histiocytes and Touton giant cells without epithelial atypia, confirming XGI. En-bloc excision was completed without radical neck dissection. Recovery was uneventful, and no recurrence was observed at 14 months. Only three adult BCC-XGI cases have been reported, including the present patient. All showed deceptively benign imaging, disproportionate intraoperative fibrosis, and reliance on frozen section to exclude malignancy. Chronic leakage or rupture of cyst contents - possibly potentiated by fine-needle aspiration - appears to trigger the xanthogranulomatous response. Surgeons should consider XGI when encountering fibrotic lateral cervical cysts in older adults. Prompt intraoperative frozen-section analysis enables conservative excision and prevents overtreatment.

CureusVol. 17(6)
Iwate Medical University (JP)
Zero hunger
Openalex Percentile: Top 84%
Infectious Disease Case Reports and Treatments
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Adult-Onset Xanthogranulomatous Inflammation Presenting as a Cervical Branchial-Cleft Cyst — Shinichi Oikawa, Ryoukichi Ikeda, et al. · Cureus (2025) | TGRS Research Map | TGRS