Xanthoma Disseminatum: Case Report and Literature Review

Pinto, Miguel E.; Escalaya, Glenda; Escalaya, María E.; Pinto, José Luı̀s; Chián, César

doi:10.4158/ep10040.cr

Cited by 16 publications

(17 citation statements)

References 13 publications

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“…PST in children has also been reported in association with other CNS malignancies and tuberculosis . Rarer causes of PST in adults, including sarcoidosis , Wegener granulomatosis , hemophagocytic lymphohistiocytosis , Behçet disease , xanthoma disseminatum , Whipple disease , or metastatic spread of non‐CNS non‐lymphoid malignancies , have not been reported in children.…”

Section: Discussionmentioning

confidence: 99%

Predictors of neoplastic disease in children with isolated pituitary stalk thickening

Robison

Prabhu

Sun

et al. 2013

Pediatric Blood & Cancer

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Section: Discussionmentioning

confidence: 99%

Predictors of neoplastic disease in children with isolated pituitary stalk thickening

Robison

Prabhu

Sun

et al. 2013

Pediatric Blood & Cancer

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“…Interestingly, however, while the skin lesions in our patient resolved completely and brain imaging showed resolution of the pituitary stalk thickening, the symptoms of diabetes insipidus and amenorrhea remained, perhaps reflecting permanent damage to the pituitary. According to the involved location, diabetes insipidus may be accompanied by varying degrees of hypopituitarism and hyperprolactinemia [15]. Our patient had vasopressin-sensitive diabetes insipidus and hypogonadotropism, but her prolactin, cortisol, and growth hormone levels were normal.…”

Section: Discussionmentioning

confidence: 92%

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 is a rare form of normolipidemic, mucocutaneous xanthoma unaccompanied by hyperlipidemia; reactive granulomatous proliferative disorder of reticuloendothelial histiocytes is considered the cause [3, 4]. The condition was first reported in 1938 by Montgomery and Osterberg, and approximately 100 cases have been reported to date [5].…”

Section: Discussionmentioning

confidence: 99%

Osteoarticular lesion in xanthoma disseminatum treated with total hip arthroplasty: a case report

et al. 2019

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Introduction Xanthoma disseminatum is a very rare disease classified as a benign non-Langerhans cell histiocytosis, which is rarely associated with osteoarticular lesions. There is only a report of tumor abrasion during treatment of osteoarticular lesions of this disease, artificial joint replacement has not been reported. We describe a patient in whom bilateral total joint replacement was performed for disseminated xanthoma lesions of the hip joints. Case presentation A 34-year-old Japanese woman had a chief complaint of bilateral coxalgia. She had been diagnosed as having disseminated xanthoma. Radiographs showed numerous 5-mm radiolucent bands that resembled worm-eaten tracks in the lower part of the femoral heads adjacent to the joint surface. In addition, short tau inversion recovery imaging scans showed high-intensity areas from the femoral head to the neck in both femurs, suggesting bone marrow edema. Total hip arthroplasty was performed for hip arthrosis on both hip joints caused by disseminated xanthoma. Deflection of the implants was a concern from the early stages postoperatively, but both the imaging and clinical findings have been satisfactory for 4 years of follow-up. Conclusions A very unusual hip joint lesion of xanthoma disseminatum was replaced with a total artificial joint replacement, and the course over 4 years was good. Our patient’s course will continue to be followed carefully.

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Xanthoma Disseminatum: Case Report and Literature Review

Cited by 16 publications

References 13 publications

Predictors of neoplastic disease in children with isolated pituitary stalk thickening

Predictors of neoplastic disease in children with isolated pituitary stalk thickening

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

Osteoarticular lesion in xanthoma disseminatum treated with total hip arthroplasty: a case report

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