A 7-Year History of Necrobiotic Xanthogranuloma following Asymptomatic Multiple Myeloma: A Case Report

Inthasotti, Siriluk; Wanitphakdeedecha, Rungsima; Manonukul, Jane

doi:10.1155/2011/927852

Cited by 16 publications

(21 citation statements)

References 27 publications

(39 reference statements)

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“…4 Hematologic disorders such as MM are a common association with NXG as seen in our patient. Internal organ involvement, however, has also been shown to occur.…”

Section: Discussionsupporting

confidence: 55%

“…2,7 NXG may involve various extracutaneous sites, including the lung, myocardium, larynx, pharynx, skeletal muscle, kidney, ovary, and intestine. 3,4 These include monoclonal gammopathy seen in most patients, most commonly IgG(k), and multiple myeloma (MM) occurring in < 10% of patients. 3,4 These include monoclonal gammopathy seen in most patients, most commonly IgG(k), and multiple myeloma (MM) occurring in < 10% of patients.…”

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confidence: 99%

“…3 Lymphoproliferative disorders are frequently associated with NXG. 3,4 Histologically, necrobiosis of collagen is seen with most cases exhibiting bandlike granulomatous inflammation alternating with inflammatory cells. 3,4 Histologically, necrobiosis of collagen is seen with most cases exhibiting bandlike granulomatous inflammation alternating with inflammatory cells.…”

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confidence: 99%

“…3 Foamy histiocytes, Touton giant cells, asteroid bodies, cholesterol clefts, and plasma cells may also be seen. 4,7 There is a case report of successful treatment with lenalidomide. Various agents including corticosteroids, melphalan, chlorambucil, methotrexate, thalidomide, cyclophosphamide, and azathioprine have been used with limited efficacy.…”

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confidence: 99%

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Successful Treatment of Necrobiotic Xanthogranuloma with Intravenous Immunoglobulin

2013

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Background: Necrobiotic xanthogranuloma (NXG) is a rare chronic disorder characterized by firm yellow to red-orange nodules and plaques affecting the face, abdomen, and extremities with the potential for systemic involvement. NXG has a close association with monoclonal gammopathies, and there is a predilection for the development of multiple myeloma. Treatment options are varied due to inconsistent results seen with the use of corticosteroids, immunomodulators, chemotherapeutic agents, and antibiotics.Objective: We describe a patient with smoldering multiple myeloma associated with progressive NXG successfully treated with high-dose intravenous immunoglobulin (IVIG).Conclusion: Our case adds to the single previous report of two cases of NXG with significant improvement from treatment with IVIG and confirms the efficacy of this treatment modality.Contexte: Le xanthogranulome né crobiotique (XN) est une affection chronique rare, qui se caracté rise par la pré sence de nodules fermes, dont la couleur varie du jaune au rouge orangé , et de plaques qui touchent la face, l'abdomen, et les membres, et il peut mê me s'é tendre à d'autres parties du corps. Le XN est en association é troite avec les gammapathies monoclonales, et il se trouve souvent en concomitance avec le myé lome multiple. Les traitements diffè rent sensiblement compte tenu de la variabilité des ré sultats obtenus avec les corticosté roïdes, les immunomodulateurs, les agents chimiothé rapeutiques, et les antibiotiques.Objectif: Il sera question, dans le pré sent article, du cas d'un patient atteint de myé lome multiple latent, en association avec un XN é volutif, traité avec succè s par des doses é levé es d'immunoglobulines (Ig) intraveineuses (i.v.).Conclusion: Cet exposé de cas s'ajoute au seul autre rapport sur le traitement de deux cas de XN par les Ig i.v., qui a permis une amé lioration sensible de l'é tat, et confirme ainsi l'efficacité du procé dé thé rapeutique.

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“…4 Hematologic disorders such as MM are a common association with NXG as seen in our patient. Internal organ involvement, however, has also been shown to occur.…”

Section: Discussionsupporting

confidence: 55%

mentioning

confidence: 99%

mentioning

confidence: 99%

mentioning

confidence: 99%

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Successful Treatment of Necrobiotic Xanthogranuloma with Intravenous Immunoglobulin

2013

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“…The term ‘necrobiotic xanthogranuloma’ was first coined by Kossard and Winkleman in 1980, when they reported eight patients with xanthomatous plaques and nodules with a predisposition for the periorbital, trunk and limb areas . NXG is a rare multisystem histiocytic disease, usually occurring during the sixth decade of life with an equal sex predilection . Common manifestations include yellow/red/brown nodules, which can develop into plaques, and approximately 43% of patients can develop ulceration and atrophy .…”

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confidence: 99%

Necrobiotic xanthogranuloma treated with lenalidomide

et al. 2017

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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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A 7-Year History of Necrobiotic Xanthogranuloma following Asymptomatic Multiple Myeloma: A Case Report

Cited by 16 publications

References 27 publications

Successful Treatment of Necrobiotic Xanthogranuloma with Intravenous Immunoglobulin

Successful Treatment of Necrobiotic Xanthogranuloma with Intravenous Immunoglobulin

Necrobiotic xanthogranuloma treated with lenalidomide

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

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