Xanthoma disseminatum and multiple myeloma

Maize, J. C.

doi:10.1001/archderm.110.5.758

Cited by 18 publications

(10 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: 75%

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: 75%

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Szalat

Arnulf

Karlin

et al. 2011

Blood

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“…In our 5 adult cases and in most of those subsequently reported in the literature [6][7][8][9][10][11][12][13][14][15][16][17][18][19][20], the average age of onset of the disease was over 40 years. At least 3 cases had their onset after the 7th decade [7,15,16], while in less than 10 cases onset was before the 5th year of life [5,…”

Section: Resultsmentioning

confidence: 91%

The Various Clinical Patterns of Xanthoma disseminatum

et al. 1995

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Background: Xanthoma disseminatum (XD) is a rare mucocutaneous xanthoma nowadays classified as a benign non-Langerhans cell histiocytosis. Objective: We studied 7 cases of XD with a long follow-up and reviewed the literature in order to compare and discuss previous reports. Methods: We studied clinical manifestations, course, histopathologic, ultrastructural and immunohistochemical findings of 7 cases of XD followed for 1–20 years. Results: We report our findings and compare our cases with those described in the literature. Conclusions: XD and Langerhans cell histiocytosis were distinguished by clinical differences, as well as ultrastructural and immunohistochemical methods. Three clinical patterns of XD appeared to emerge in our group of patients and in the cases reviewed from the literature.

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“…It is a rare normolipidemic mucocutaneous xanthomatosis that arises from the proliferation of non-Langerhans cell histiocytes. Age at onset ranges from as early as 8 months [10] to 85 years [11]. Altman and Winkelmann [3] reported that two thirds of the cases occurred before 25 years of age, but according to Caputo et al [4], the average age at disease onset was over 40 years.…”

Section: Discussionmentioning

confidence: 99%

“…Our patient had vasopressin-sensitive diabetes insipidus and hypogonadotropism, but her prolactin, cortisol, and growth hormone levels were normal. There have also been reports of rare associations of XD with multiple myeloma, Waldenström’s macroglobulinemia, hypothyroidism, and hyperthyroidism [4,11,16,17]. …”

Section: Discussionmentioning

confidence: 99%

A Case of Xanthoma Disseminatum with Spontaneous Resolution over 10 Years: Review of the Literature on Long-Term Follow-Up

Park¹,

Joe²,

Kang³

et al. 2011

Dermatology

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Xanthoma disseminatum (XD) is a rare and potentially progressive non-Langerhans-cell histiocytosis. To date, a few cases of XD with spontaneous complete resolution have been described. The present report describes a 16-year-old girl who presented with yellow to red-brown papules and nodules on her eyelids, cheeks, axillae, back and buttocks. Indirect laryngoscopy showed multiple xanthomatous plaques on the larynx, posterior pharynx, epiglottis, and vocal cords. Additional findings were polyuria, polydipsia, and amenorrhea. Skin biopsy and electron microscopy results confirmed the diagnosis of XD. The patient was treated with fenofibrate, simvastatin, desmopressin, and sex-hormone replacement therapy. Her skin lesions began to slowly fade 6 years after disease onset, eventually resolving spontaneously and completely, but leaving an atrophic scar, frank anetoderma, and persisting diabetes insipidus. This case report together with a review of the English-language literature on the long-term follow-up of XD patients provides additional information on the natural history of this disease.

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Xanthoma disseminatum and multiple myeloma

Cited by 18 publications

References 0 publications

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

Pathogenesis and treatment of xanthomatosis associated with monoclonal gammopathy

The Various Clinical Patterns of Xanthoma disseminatum

A Case of Xanthoma Disseminatum with Spontaneous Resolution over 10 Years: Review of the Literature on Long-Term Follow-Up

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