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doi: https://doi.org/10.4132/jptm.2019.01.08    [Epub ahead of print]
Primary Necrobiotic Xanthogranulomatous Sialadenitis with Submandibular Gland Localization without Skin Involvement: A Case Report and Review
Myunghee Kang1, Na Rae Kim1, Dong Hae Chung1, Jae Yeon Seok1, Dong Young Kim2
1Department of Pathology, Gachon University Gil Medical Center, Gachon University College of Medicine, Incheon, Korea
2Department of Otorhinolaryngology-Head and Neck Surgery, Gachon University Gil Medical Center, Gachon University College of Medicine, Incheon, Korea
Corresponding Author: Na Rae Kim ,Tel: +82 32 460 3073, Fax: +82 32 460 2394, Email: clara_nrk@gilhospital.com
Received: November 26, 2018;  Revised: December 28, 2018  Accepted: January 8, 2019.  Published online: January 16, 2019.
Necrobiotic xanthogranulomatous reaction is a multiorgan, non-Langerhans cell histiocytosis with an unknown etiology. Occurrence in the salivary gland is extremely rare. We recently identified a case of necrobiotic xanthogranulomatous sialadenitis in a 73-year-old Korean woman who presented with a painless palpable lesion in the chin. There was no accompanying cutaneous lesion. Partial resection and subsequent wide excision with neck dissection were performed. Pathologically, examination showed a severe inflammatory lesion that included foamy macrophages centrally admixed with neutrophils, eosinophils, lymphocytes, plasma cells, and scattered giant cells, as well as necrobiosis. During the 7-month postoperative period, no grossly remarkable change in size was noted. Necrobiotic xanthogranulomatous inflammation may be preceded by or combined with hematologic malignancy. Although rare, clinicians and radiologists should be aware that an adhesive necrobiotic xanthogranulomatous in the salivary gland may present with a mass-like lesion. Further evaluation for hematologic disease and close follow-up are needed when a pathologic diagnosis is made.
Key Words: Necrobiotic xanthogranuloma; Salivary glands; Sialadenitis; Hematologic neoplasms
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