J Pathol Transl Med.  2019 Jul;53(4):261-265. 10.4132/jptm.2019.01.08.

Primary Necrobiotic Xanthogranulomatous Sialadenitis with Submandibular Gland Localization without Skin Involvement

• Kang M
• Kim NR
• Chung DH
• Seok JY
• Kim DY

• KMID: 2454606
• DOI: http://doi.org/10.4132/jptm.2019.01.08

Abstract

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. Pathological 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 12-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 xanthogranuloma 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.

Keyword

Necrobiotic xanthogranuloma; Salivary glands; Sialadenitis; Hematologic neoplasms

MeSH Terms

Adhesives
Aged
Chin
Diagnosis
Eosinophils
Female
Follow-Up Studies
Giant Cells
Hematologic Diseases
Hematologic Neoplasms
Histiocytosis
Humans
Inflammation
Lymphocytes
Macrophages
Neck Dissection
Necrobiotic Disorders
Necrobiotic Xanthogranuloma
Neutrophils
Plasma Cells
Postoperative Period
Salivary Glands
Sialadenitis*
Skin*
Submandibular Gland*
Adhesives

Figure

• 

  Fig. 1. Necrobiotic xanthogranulomatous sialadenitis. (A) Head and neck computed tomography shows several conglomerated masses in the submandibular gland. (B) Fine needle aspiration cytology reveals many lymphocytes and scattered histiocytes with tingible bodies (PAP). (C) Gross picture of the resected specimen shows a well-circumscribed, ovoid, white-tan mass with yellowish necrotic foci. (D) Necrobiotic xanthogranuloma shows a zone of thick collagen surrounded by lymphohistiocytic palisading granulomas. (E) Foreign body type and Touton-type giant cells are noted (CD68 immunostaining, inset). (F) Cholesterol clefts (upper center) and foam cells are seen adjacent to necrobiosis.

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