Plane xanthoma and multiple myeloma with lipoprotein--paraprotein complexing

Taylor, J. S.

doi:10.1001/archderm.114.3.425

Cited by 24 publications

(23 citation statements)

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“…However, Touton cells have been observed in HX and NX in several cases in the literature. 20,49,57,62,[78][79][80][81][82][83][84][85] Moreover, as demonstrated here, foam cells and Touton cells share phenotypic characteristics, including the surface expression of the CD163 antigen, which would suggest a common macrophagic origin. Finally, immune complex formation resulting from antibody activity of the MIg directed against various lipoproteins appears to represent a common physiopathologic pathway leading to a lipid accumulation in the macrophagic cells.…”

Section: Discussionmentioning

confidence: 76%

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: 76%

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Szalat

Arnulf

Karlin

et al. 2011

Blood

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“…A predominant association of DPNX with paraproteinemia of light-chain immunoglobulins seems to occur with the λ isotype prevailing. 8,9 As in our case, an extensive cutaneous involvement is in favor of an underlying systemic disease; multiple myeloma as well as other hematological, lymph proliferative disorders were ruled out and a MGUS was considered the An Bras Dermatol. 2011;86(4Supl1):S50-2.…”

Section: Discussionmentioning

confidence: 64%

“…Those complexes may be recognized as modified LDL by scavenger macrophages leading to development of cutaneous xanthomas. 7,8 For this reason, DPNX is considered a histiocytosisderived xanthomatosis (histiocytic lipidization). 5 Since the first time that the association between paraproteins and DPNX has been suggested, 21 cases with multiple myeloma and xanthomatosis have been confirmed as having lipoprotein-paraprotein complexes.…”

Section: Discussionmentioning

confidence: 99%

“…5 Since the first time that the association between paraproteins and DPNX has been suggested, 21 cases with multiple myeloma and xanthomatosis have been confirmed as having lipoprotein-paraprotein complexes. 8 The deposition of the complexes seems to be influenced more by the properties of the complexes rather than the lipid levels, explaining why most of the patients have normal serum lipid levels. 10,11 Histopathological examination shows histiocytic foamy cells diffusely spread in the upper dermis with and an admixture of histiocytes and lymphocytes.…”

Section: Discussionmentioning

confidence: 99%

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Diffuse plane xanthomatosis associated with monoclonal gammopathy

Rosmaninho

Fernandes

Guimas

et al. 2011

An. Bras. Dermatol.

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Resumo: A xantomatose plana difusa normolipêmica (XPDN) é uma dermatose adquirida rara, muitas vezes associada a doenças sistêmicas, nomeadamente neoplasias hematológicas(sobretudo o mieloma múl-tiplo) ou a processos linfoproliferativos. A XPDN pode preceder o aparecimento dessas doenças em vários anos, sendo por isso recomendada uma vigilância clínica e laboratorial periódica, mesmo para os doentes que aparentemente não apresentam uma doença associada. Descrevemos um caso associado à gamopatia monoclonal. Este caso demonstra a importância das manifestações cutâneas como primeira manifestação de doenças hematológicas importantes e por isso os clínicos devem estar familiarizados com esta entidade. Palavras-chave: Mieloma múltiplo; Paraproteinemias; Xantomatose Abstract: Diffuse plane normolipemic xanthomatosis (DPNX) is a rare, non-inherited disease that is often associated with systemic diseases, mainly malignant hematological (especially multiple myeloma) or lymph proliferative disorders. The DPNX can precede the appearance of such conditions by several years, so careful follow-up and periodic laboratory examinations are recommended even for patients that seemed to have no underlying disease. We describe a case associated with monoclonal gammopathy. This case shows that dermatological lesions can be the first manifestation of important hematological diseases and so physicians should be familiarized with this entity.

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“…Cutaneous xanthoma occurring in association with paraproteinemia is a well-documented clinical abnormality that is seen in patients with multiple myeloma, benign monoclonal gammapathies, and other reticular endothelial disorders (1)(2)(3)(4)(5)(6). In many of these individuals the occurrence of xanthoma is associated with hyperlipidemia, but it should be recognized that cutaneous xanthoma in association with paraproteinemia can be observed in individuals whose plasma lipid levels are within the normal range (1,3,5,(7)(8)(9).…”

Section: Introductionmentioning

confidence: 99%

Cutaneous xanthoma in association with paraproteinemia in the absence of hyperlipidemia.

Feingold¹,

Castro²,

Ishikawa³

et al. 1989

J. Clin. Invest.

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In the present report we describe a patient with multiple myeloma and long-standing paraproteinemia who developed xanthoma in the absence of an elevation in plasma cholesterol or triglyceride concentrations. Studies demonstrated that our patient's monoclonal IgG antibody interacted with apoprotein B-100. The LDL-antibody complex isolated from our patient did not affect the degradation of LDL by human fibroblasts, indicating that while IgG derived from our patient interacted with LDL it did not alter the metabolism of this lipoprotein by the LDL receptor pathway. Since the LDL receptor pathway is the major route of LDL metabolism, this probably explains why our patient was not hyperlipidemic. In contrast to an absence of effect on the LDL receptor, our patient's LDL-antibody complex stimulated cholesterol esterification within macrophages indicating the uptake and degradation of the LDLantibody complex. The LOL-antibody complex inhibited the degradation of acetyl LDL by macrophages (scavenger pathway), demonstrating that our patient's LDL-antibody complex was recognized as a modified LDL. Moreover, mixing Ig from our patient with normal LDL also resulted in the normal LDL increasing the esterification of cholesterol by macrophages.One can hypothesize that our patient's monoclonal IgG-LDL complex interacted with the macrophage scavenger receptor, thereby resulting in the occurrence of xanthoma in the absence of hyperlipidemia.

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Plane xanthoma and multiple myeloma with lipoprotein--paraprotein complexing

Cited by 24 publications

References 0 publications

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Diffuse plane xanthomatosis associated with monoclonal gammopathy

Cutaneous xanthoma in association with paraproteinemia in the absence of hyperlipidemia.

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