Necrobiotic xanthogranuloma with multiple myeloma. Case report and pathogenic hypotheses

Langlois, Sandrine; Brochot, P.; Réguiai, Z.; Bernard, Philippe; Moreau, Eric; Gagneux-Lemoussu, Laurence; Eschard, J. P.; Étienne, Jean Claude

doi:10.1016/j.jbspin.2005.03.008

Cited by 15 publications

(15 citation statements)

References 7 publications

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“…First, HX and NX lesions can coexist with lesions of NXG in the same patient as seen both in patients from our series and from the literature. 46,[64][65][66][67] Second, the 3 forms share common histologic features, namely, the presence of foam cells and Touton cells. The latter is typically considered as characteristic of NXG; and, indeed, we did not find such cells in any of our patients with HX and NX.…”

Section: Discussionmentioning

confidence: 99%

“…In 5 patients, xanthoma and NXG were present at the same time. 46,[64][65][66][67] Extracutaneous lesions were reported in 23 cases (17.6%), including 6 patients with several localizations. They were pulmonary (n ϭ 7), 8 cardiac (n ϭ 6), 8,37 oral cavity (n ϭ 4), 38,65 orbital (n ϭ 3), 8 hepatic (n ϭ 2), 8,37 spleen (n ϭ 2), 8 medullar (n ϭ 1), 68 genital (penis; n ϭ 1), 34 mastoid (n ϭ 1), 32 facial nerve (n ϭ 1), 32 and renal (n ϭ 1).…”

Section: Comparison With Existing Literaturementioning

confidence: 99%

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Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Szalat

Arnulf

Karlin

et al. 2011

Blood

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Xanthomas are a common manifestation of lipid metabolism disorders. They include hyperlipemic xanthoma, normolipemic xanthoma, and a related condition, necrobiotic xanthogranuloma (NXG). All 3 forms can be associated with monoclonal immunoglobulin (MIg). In an attempt to improve diagnosis, understanding, and treatment of this association, we retrospectively analyzed a personal series of 24 patients (2 hyperlipemic xanthoma, 11 normolipemic xanthoma, and 11 NXG) and 230 well-documented reports from the literature. With the exception of the nodules and plaques featured in NXG, the clinical presentation of xanthomatous lesions usually resembled that seen in common hyperlipidemic forms and could not be used to suspect MIg-associated xanthomas. Extracutaneous sites were not rare. The MIg was an IgG in 80% of cases. Myeloma was diagnosed in 35%. Hypocomplementemia with low C4 fraction was present in 80% of studied patients. Low C1 inhibitor serum levels were found in 53%. Cryoglobulinemia was detected in

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Section: Discussionmentioning

confidence: 99%

Section: Comparison With Existing Literaturementioning

confidence: 99%

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Szalat

Arnulf

Karlin

et al. 2011

Blood

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“…In addition, several patients carried myeloproliferative or lymphoproliferative diagnoses, although a bone marrow biopsy was not reported. These included a patient with non‐Hodgkin's lymphoma, 52 a patient with chronic lymphocytic leukemia, 52 one with Hodgkin's lymphoma, 27 two patients with monoclonal gammopathy of undetermined significance, 42,52 a patient with lymphoplasmacytic lymphoma, 29 one patient with “smoldering myeloma”, 34 and a patient with multiple myeloma 53 …”

Section: Bone Marrow Findingsmentioning

confidence: 99%

“…6,12,42 Melphalan was used more than any other alkylating agent. Ten of 12 patients improved with melphalan, 9,14,17,30,31,40,42,50,53 although one patient with multiple myeloma later showed recurrence of skin lesions, 44 and the duration of the remaining responses is unknown.…”

Section: Treatmentmentioning

confidence: 99%

Necrobiotic xanthogranuloma

and

Mehregan

2008

Int J Dermatology

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“…It was hypothesized that lipid-laden monocytes and immunoglobulins are deposited in the skin and elicited a giant cell inflammatory reaction [3]. On the contrary, M-protein may be the primary abnormality, and skin lesions arise from secondary proliferation of macrophages bearing receptors for Fc portion of their M-protein [4]. Another study identified spirochaetal organism in NXG lesions, suggesting infectious etiology of NXG [5].…”

Section: Discussionmentioning

confidence: 99%

Periorbital necrobiotic xanthogranuloma without paraproteinemia

Nakamura-Wakatsuki¹,

Yamamoto²

2013

Our Dermatol Online

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Introduction: Necrobiotic xanthogranuloma (NXG) is a rare histiocytic disease which most frequently involves periorbital areas. NXG is associated with paraproteinemia with a ratio over 80%. However, a few cases of NXG without paraproteinemia (isolated NXG) have also been reported. Main observation: A 58-year-old Japanese woman complained about asymptomatic dermal nodules on the bilateral lower eyelids. The histopathological examination revealed granulomatous inflammation. Paraproteinemia was not detected either in the serum or in the urine. The size of the nodules has been constant, and she has been under careful follow-up without any systemic therapies. Conclusions: NXG is frequently associated with paraproteinemia, which may develop later. Patients with NXG need careful follow-up including repeated blood tests.

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Necrobiotic xanthogranuloma with multiple myeloma. Case report and pathogenic hypotheses

Cited by 15 publications

References 7 publications

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Necrobiotic xanthogranuloma

Periorbital necrobiotic xanthogranuloma without paraproteinemia

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