Journal of Pathology and Translational Medicine

Case Study

Primary Necrobiotic Xanthogranulomatous Sialadenitis with Submandibular Gland Localization without Skin Involvement: Myunghee Kang, Na Rae Kim, Dong Hae Chung, Jae Yeon Seok, Dong Young Kim; J Pathol Transl Med. 2019;53(4):261-265. Published online January 16, 2019; DOI: https://doi.org/10.4132/jptm.2019.01.08

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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.

Citations

Citations to this article as recorded by

Five Cases of Xanthogranulomatous Sialadenitis
Satoshi Kiyama, Hiroyuki Iuchi, Kotoko Ito, Kengo Nishimoto, Tsutomu Matsuzaki, Masaru Yamashita
Practica Oto-Rhino-Laryngologica.2022; 115(4): 315. CrossRef
Xanthogranulomatous change in a pleomorphic adenoma: An extremely rare variant/degenerative change. Is it fine needle aspiration induced?
Mukta Pujani, Dipti Sidam, Kanika Singh, Aparna Khandelwal, Khushbu Katarya
Diagnostic Cytopathology.2021;[Epub] CrossRef
A Case of Xanthogranulomatous Sialadenitis with Facial Palsy Mimicking Malignancy
Sang Hyun Kim, Sun Woo Kim, Sang Hyuk Lee
Korean Journal of Otorhinolaryngology-Head and Neck Surgery.2021; 64(6): 422. CrossRef
Xanthogranulomatous Sialadenitis, an Uncommon Reactive Change is Often Associated with Warthin’s Tumor
Lihong Bu, Hui Zhu, Emilian Racila, Sobia Khaja, David Hamlar, Faqian Li
Head and Neck Pathology.2020; 14(2): 525. CrossRef
A Case of Xanthogranulomatous Sialadenitis of the Sublingual Gland:A Review of Literature
Naoya KITAMURA, Seiji OHNO, Tetsuya YAMAMOTO
Journal of Japanese Society of Oral Medicine.2019; 25(1): 20. CrossRef

Case Reports

IgG4-Related Sclerosing Sialadenitis: Report of Three Cases.: Ji Seon Bae, Joo Young Kim, Sang Hak Han, Seung Ho Choi, Kyung Ja Cho; Korean J Pathol. 2011;45:S36-S40.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S36

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Abstract PDF

Chronic sclerosing sialadenitis, Mikulicz disease or Kuttner tumor has been recently recognized as a spectrum of IgG4-related sclerosing disease. IgG4-related disease is characterized by a high serum IgG4 level and tissue infiltration of IgG4-positive plasmacytes. We report three cases of chronic sclerosing sialadenitis with variably associated systemic involvement. All patients presented with a submandibular mass or swelling, and all the resected submandibular glands showed diffuse lymphocytic infiltration, lymphoid follicles, and septal fibrosis. Two of the specimens revealed numerous IgG-positive plasma cells, most of which were IgG4-positive on immunohistochemical staining. One of them was associated with dacryoadenitis and hypophysitis. The other patient had ureterorenal lesions. Immunohistochemical study was unavailable in remaining one case, but the histologic features along with elevated IgG level and associated pancreatitis supported the diagnosis. All patients received steroid therapy postoperatively and are doing well. Salivary gland involvement in IgG4-related fibrosclerosis should be recognized in systemic medical pathology.

Citations

Citations to this article as recorded by

A Case of Immunoglobulin G4-Related Sialadenitis and Dacryoadenitis
Kyoung Kyu Lee, Hun Po Cho, Young Min Lee, Jun Ho Park
Korean Journal of Otorhinolaryngology-Head and Neck Surgery.2013; 56(7): 444. CrossRef

Fine Needle Aspiration Cytology of Chronic Sclerosing Sialadenitis with Mucinous Metaplasia in Parotid Gland: A Case Report.: Jae Yeon Seok, Woo Hee Jung, Xu Xiang Fan, Jin Kim, Soon Won Hong; Korean J Cytopathol. 2005;16(2):102-105.

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Abstract PDF: Chronic sclerosing sialadenitis, also known as Kuttner tumor, is a benign chronic inflammatory lesion of the salivary gland. Here, we describe a case of chronic sclerosing sialadenitis with mucinous ductal metaplasia in a parotid gland, which was confused with low-grade mucoepidermoid carcinoma on aspiration cytology.

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