Plane Xanthomatosis With Antilipoprotein Autoantibody

Kodama, Hajime; Nakagawa, Shojiro; Tanioku, K

doi:10.1001/archderm.1972.01620080048014

Cited by 48 publications

(6 citation statements)

References 20 publications

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“…Other authors postulated that many paraproteins display antilipoprotein activity and form complexes with lipoproteins that are deposited in the dermis. [13] Williford et al [14] considered NPX to be a histiocytosis-derived xanthomatosis in the spectrum of non-X histiocytosis. Recently reported cases have shown that diffuse plane xanthoma can also occur in previously inflamed skin disorders, such as atopic dermatitis and erythroderma.…”

Section: Discussionmentioning

confidence: 99%

Cutaneous Normolipemic Plane Xanthoma with Supraglottic Involvement in a Patient with Hand-Schüller-Christian Disease

Chen

Chung

et al. 2009

American Journal of Clinical Dermatology

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Normolipemic plane xanthoma (NPX) is a histiocytic disorder characterized by yellow-orange plaques in the periorbital areas, neck, upper trunk, and flexural folds. Association with systemic disease or paraproteinemia has been reported previously, but rarely with Langerhans cell histiocytosis (LCH). We report a case of Hand-Schüller-Christian disease (a type of LCH) in a patient who developed NPX with supraglottic involvement. NPX developed after several courses of chemotherapy and the supraglottic xanthoma occurred about 2 years later. The coexistence of LCH and non-LCH histiocytic lesions in this patient could be a result of chemotherapy-induced changes or may be just coincidental.

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Section: Discussionmentioning

confidence: 99%

Cutaneous Normolipemic Plane Xanthoma with Supraglottic Involvement in a Patient with Hand-Schüller-Christian Disease

Chen

Chung

et al. 2009

American Journal of Clinical Dermatology

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“…According to the published work, 12,13 DPNX associated with monoclonal gammopathy may produce lipid abnormalities by a specific interaction between a monoclonal immunoglobulin and lipoprotein metabolism. It is postulated that paraprotein‐lipoprotein complexes (IgG‐LDL) may cause a perivascular deposition that is scavenged by macrophages, finally resulting in the development of cutaneous xanthomas 14 .…”

Section: Discussionmentioning

confidence: 99%

A case of diffuse plane normolipemic xanthomatosis associated with pancytopenia and monoclonal gammopathy

Kourou

Suga

Muramatsu

et al. 2006

The Journal of Dermatology

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We report a case of diffuse plane normolipemic xanthomatosis (DPNX) which showed poorly demarcated, uncommon, yellow macules symmetrically distributed on the nape, axillae and inguinal folds accompanied by severe, persistent itching. Histopathological and ultrastructural studies of skin biopsy specimens revealed the existence of some foamy cells and the deposition of neutral fat in the upper papillary dermis. Laboratory investigations and bone marrow aspirate smears showed that our patient had myelodysplastic syndrome (MDS) associated with pancytopenia and monoclonal gammopathy of undetermined significance. Because our patient had neither a malignant hematological disorder nor a severe systemic disease, monoclonal gammopathy might explain the pathogenesis of DPNX in the present case.

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“…Although type III hyperlipoproteinemia usually results from an inherited defect in the composition of Apo-E, an acquired form of this disease has been reported to be associated with myeloma and immunoglobulin-lipoprotein complexes. 8,10,[17][18][19][20][21][22][23][24][25] Studies 11,26,27 have shown that direct interaction of immunoglobulin with lipoproteins appear to disrupt normal lipoprotein metabolism, thus, leading to hyperlipidemia. Indeed, immunostaining studies confirmed the presence of IgA antibodies complexed to the patient's VLDL fraction.…”

Section: Discussionmentioning

confidence: 99%