Necrobiotic xanthogranuloma of the parotid gland

Zainal, A; Razif, M Y Mohd; Makhashen, Maraei Bin; Swaminathan, Manickam; Mazita, A

doi:10.1017/s0022215109991563

Cited by 5 publications

(5 citation statements)

References 13 publications

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“…But in our case first-time FNAC did and found Xanthogranulomatous inflammation on cytology. Some studies described XG inflammation associated with Warthin's tumour [2][3]6]. In our case aetiology not clear for XG inflammation.…”

Section: Malek Et Al Reported a Series Of 26 Patients Withcontrasting

confidence: 58%

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Xanthogranulomatous Inflammation of Salivary Gland Diagnosed By Fine Needle Aspiration Cytology- A Rare Case Report

Anju¹

2021

Prog Med Sci

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Section: Malek Et Al Reported a Series Of 26 Patients Withcontrasting

confidence: 58%

“…It commonly seen in kidney, causative agents exact not known, XGI believed to develop in the setting of a suppurative infection with renal outflow obstruction [1]. Mostly studies demonstrated XG inflammation in parotid gland with slow-growing mass [2][3].…”

Section: Introductionmentioning

confidence: 99%

Xanthogranulomatous Inflammation of Salivary Gland Diagnosed By Fine Needle Aspiration Cytology- A Rare Case Report

Anju¹

2021

Prog Med Sci

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“…In the systematic review, the initial search yielded 1199 records, 151 of which were included (Figure). These reports described 201 patients with NXG, with demographics, comorbidities, and clinical features (detailed in the eTable in the Supplement).…”

Section: Resultsmentioning

confidence: 99%

A Multicenter Cross-Sectional Study and Systematic Review of Necrobiotic Xanthogranuloma With Proposed Diagnostic Criteria

et al. 2020

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IMPORTANCE Necrobiotic xanthogranuloma (NXG) is a non-Langerhans cell histiocytosis classically associated with paraproteinemia attributable to plasma-cell dyscrasias or lymphoproliferative disorders. Despite the morbidity of NXG, the literature is limited to case reports and small studies, and diagnostic criteria are lacking.OBJECTIVE To evaluate the characteristics of NXG and propose diagnostic criteria. DESIGN, SETTING, AND PARTICIPANTSThis multicenter cross-sectional study was conducted at tertiary academic referral centers and followed by a systematic review and a consensus exercise. The multicenter cohort included patients with NXG diagnosed at the Brigham and Women's and Massachusetts General Hospitals (2000-2018), the University of Iowa Hospitals and Clinics (2000-2018), and the University of Pennsylvania Health System (2008)(2009)(2010)(2011)(2012)(2013)(2014)(2015)(2016)(2017)(2018). The systematic review was conducted in 2018 and included patients with NXG identified in the Cochrane, Ovid EMBASE, PubMed, and Web of Science databases. The consensus exercise was conducted by 8 board-certified dermatologists to identify diagnostic criteria.MAIN OUTCOMES AND MEASURES Demographic factors, comorbidities, clinical features, and treatment response. RESULTSOf 235 included patients with NXG (34 from the multicenter cohort and 201 from the systematic review results), the mean (SD) age at presentation was 61.6 (14.2) years; 147 (62.6%) were female. Paraproteinemia was detected in 193 patients (82.1%), most often IgG-κ (117 patients [50.0%]). A malignant condition was detected in 59 patients (25.1%), most often multiple myeloma (33 patients [14.0%]). The overall rate of paraproteinemia and/or a malignant condition was 83.8% (197 patients). In the multicenter cohort, evolution of paraproteinemia into multiple myeloma was observed up to 5.7 years (median [range], 2.4 [0.1-5.7] years) after NXG presentation. Cutaneous lesions consisted of papules, plaques, and/or nodules, typically yellow or orange in color (113 of 187 [60.4%]) with a periorbital distribution (130 of 219 [59.3%]). The eye was the leading site of extracutaneous involvement (34 of 235 [14.5%]). In the multicenter cohort, intravenous immunoglobulin had the best treatment response rate (9 of 9 patients [100%]), followed by antimalarial drugs (4 of 5 patients [80%]), intralesional triamcinolone (6 of 8 patients [75%]), surgery (3 of 4 patients [75%]), chemotherapy (8 of 12 patients [67%]), and lenalidomide or thalidomide (5 of 8 patients [63%]). The consensus exercise yielded 2 major criteria, which were (1) clinical and (2) histopathological features consistent with NXG, and 2 minor criteria, consisting of (1) paraproteinemia, plasma-cell dyscrasia, and/or other associated lymphoproliferative disorder and (2) periorbital distribution of cutaneous lesions. In the absence of foreign body, infection, or another identifiable cause, fulfillment of both major and at least 1 minor criterion were proposed to establish the diagnosis of NXG.CONCLUSIONS AND RELEVAN...

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“…Common sites include the periocular area and thorax [1]. NXG is rare in the head and neck area, and there has been only one case reported in the salivary gland [4]. NXG has characteristic histologic features, showing clusters of foamy xanthomatous macrophages, granulomas, and giant cells of foreign body type or Touton-type, cholesterol clefts, and necrobiosis [2].…”

Section: Discussionmentioning

confidence: 99%

“…The clinical course of NXG is variable and unpredictable, and recurrence of the lesion and/or multiple dissemination have been reported at other sites [13,14]. Other cases have shown systemic involvement, including hematologic malignancy and cardiac myopathy [1,4,13]. About 20% of patients in a previous study developed hematologic malignancy during the 11-year follow-up period [15].…”

Section: Discussionmentioning

confidence: 99%

Primary Necrobiotic Xanthogranulomatous Sialadenitis with Submandibular Gland Localization without Skin Involvement

Kang

Kim

Chung

et al. 2019

J Pathol Transl Med

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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.

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Necrobiotic xanthogranuloma of the parotid gland

Cited by 5 publications

References 13 publications

Xanthogranulomatous Inflammation of Salivary Gland Diagnosed By Fine Needle Aspiration Cytology- A Rare Case Report

Xanthogranulomatous Inflammation of Salivary Gland Diagnosed By Fine Needle Aspiration Cytology- A Rare Case Report

A Multicenter Cross-Sectional Study and Systematic Review of Necrobiotic Xanthogranuloma With Proposed Diagnostic Criteria

Primary Necrobiotic Xanthogranulomatous Sialadenitis with Submandibular Gland Localization without Skin Involvement

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